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Uncovering the rate and risk factors of intrathecal baclofen pump-associated complications in the adult population. 揭示成人鞘内巴氯芬泵相关并发症的发生率和风险因素。
IF 3.3 2区 医学 Q2 CLINICAL NEUROLOGY Pub Date : 2024-06-01 DOI: 10.3171/2024.3.FOCUS2471
Jakov Tiefenbach, Andre G Machado, Francois Bethoux

Objective: The aim of this study was 1) to describe the rate of intrathecal baclofen (ITB)-associated complications at a large tertiary center, and 2) to evaluate the impact of patient-related factors on the likelihood of developing such complications.

Methods: A retrospective single-center study was carried out. A total of 301 eligible patients were included in the analysis. Univariate regression models were used to evaluate the impact of age, sex, diagnosis, ambulation status, modified Ashworth scale score, body mass index, diabetes status, and pain level on the likelihood of developing a device-related infection, pump malfunction, catheter malfunction, and other clinically significant complications.

Results: Overall, 27% of patients experienced an ITB-related complication. The most common complications included infection (6%, 18/301), pump malfunction (7.3%, 22/301), and catheter malfunction (14%, 42/301). The univariate analyses revealed that the patient's ambulatory status had a significant impact on the likelihood of developing a catheter-related malfunction. Furthermore, a trend toward significance was identified between patients' preoperative body mass index and device-related infection. Finally, the risk of suffering any ITB-related complications was statistically correlated with the number of years that had passed since the initial pump implantation.

Conclusions: The authors' analysis reveals a previously underrecognized association between ambulatory status at the time of ITB pump implantation and the incidence of catheter-related complications, and confirms the impact of time since surgery on the risk of developing any ITB-related complication. The patient's age, sex, diagnosis, diabetes status, or pain level at baseline were not associated with the risk of complications. Collectively, these insights contribute novel information to the existing literature, providing practical value for physicians in guiding patient selection for ITB therapy.

研究目的本研究的目的是:1)描述一家大型三级中心的鞘内巴氯芬(ITB)相关并发症的发生率;2)评估患者相关因素对发生此类并发症的可能性的影响:方法:开展了一项回顾性单中心研究。共有 301 名符合条件的患者被纳入分析。采用单变量回归模型评估了年龄、性别、诊断、行走状态、改良阿什沃斯量表评分、体重指数、糖尿病状态和疼痛程度对发生设备相关感染、泵故障、导管故障和其他临床重大并发症的可能性的影响:总体而言,27%的患者出现了与 ITB 相关的并发症。最常见的并发症包括感染(6%,18/301)、泵故障(7.3%,22/301)和导管故障(14%,42/301)。单变量分析显示,患者的卧床状态对发生导管相关故障的可能性有显著影响。此外,还发现患者术前体重指数与设备相关感染之间存在显著性趋势。最后,发生任何 ITB 相关并发症的风险与首次植入泵后的年数在统计学上存在相关性:作者的分析揭示了在植入 ITB 泵时的卧床状态与导管相关并发症的发生率之间存在着一种以前未被充分认识到的关联,并证实了手术后的时间对发生任何 ITB 相关并发症的风险的影响。患者的年龄、性别、诊断、糖尿病状态或基线疼痛程度与并发症风险无关。总之,这些见解为现有文献提供了新的信息,为医生指导患者选择 ITB 治疗提供了实用价值。
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引用次数: 0
Palliative single-level selective dorsal rhizotomy for children with spastic cerebral palsy Gross Motor Function Classification System level IV and V: a case series and systematic review of the literature. 痉挛性脑瘫儿童大运动功能分级系统 IV 级和 V 级姑息性单层选择性背根切断术:病例系列和文献系统回顾。
IF 3.3 2区 医学 Q2 CLINICAL NEUROLOGY Pub Date : 2024-06-01 DOI: 10.3171/2024.3.FOCUS2478
Maria Licci, Nicole Alexandra Frank, Ladina Greuter, Abeelan Rasadurai, Isabel Fernandes Arroteia, Stephanie Juenemann, Raphael Guzman, Jehuda Soleman

Objective: Single-level selective dorsal rhizotomy (SDR), typically indicated for ambulatory patients, is a controversial topic for severe spastic cerebral palsy (CP) with Gross Motor Function Classification System (GMFCS) level IV or V. The objective of this case series and systematic literature review was to outline the indication and outcome of palliative SDR for nonambulatory patients with CP and GMFCS level IV and V, focusing on improvement of spasticity and of patient and caregiver reported quality of life assessment.

Methods: A retrospective case series of patients with CP and GMFCS level IV or V who underwent single-level SDR at the authors' institution is presented. Furthermore, two databases (PubMed and Embase) were searched and a systematic review with a search string based on the terms "selective dorsal rhizotomy," "cerebral palsy," and "outcome" was conducted. The primary outcome was the reduction of spasticity based on the modified Ashworth scale (MAS). Secondary outcomes were change on the Gross Motor Function Measure-66 (GMFM-66), evaluation of patient-reported outcome measures (PROMs), surgical morbidity, and mortality.

Results: Eleven consecutive children under the age of 25 years undergoing palliative single-level SDR were included. All patients showed a reduction in MAS score (mean 1.09 ± 0.66 points) and no surgical morbidity and mortality occurred. For the systematic review results from our case series, in addition to 4 reports, 274 total patients were included. Reduction of spasticity based on MAS score was noted in all studies (mean range 1.09-3.2 points). Furthermore, in 2 studies spasticity of the upper extremities showed a MAS score reduction as well (range 1.7-2.8 points). The GMFM-66 score improved in 72% of the patients, while bladder function improved in 78% of the patients. Based on the PROMs, 92% of the patients/caregivers were satisfied with the outcome and their quality of life after the procedure. Two wound infections (2.7%) and one CSF leak (1.3%) occurred, while no surgery-related deaths were described.

Conclusions: This analysis showed an improvement in spasticity, daily care, and comfort for patients with CP and GMFCS levels IV and V. Larger cohorts analyzing the outcome of palliative single-level SDR, based on the MAS, GMFM-66, and PROMs, are still needed and should be the focus of future studies. Systematic review registration no.: CRD42024495762 (https://www.crd.york.ac.uk/prospero/).

目的:单水平选择性背根切断术(SDR)通常适用于行动自如的患者,但对于粗大运动功能分级系统(GMFCS)IV级或V级的重度痉挛性脑瘫(CP)患者来说,SDR却是一个有争议的话题。本病例系列和系统性文献综述旨在概述对GMFCS IV级和V级的非行动自如的CP患者实施姑息性SDR的适应症和结果,重点关注痉挛的改善以及患者和护理人员报告的生活质量评估:方法:本文介绍了在作者所在机构接受单水平 SDR 治疗的 CP 和 GMFCS IV 级或 V 级患者的回顾性病例系列。此外,还检索了两个数据库(PubMed 和 Embase),并以 "选择性背侧根切术"、"脑性瘫痪 "和 "结果 "为检索字符串进行了系统综述。主要研究结果是根据改良阿什沃斯量表(MAS)得出的痉挛减轻程度。次要结果是粗大运动功能测量-66(GMFM-66)的变化、患者报告结果评估(PROMs)、手术发病率和死亡率:共纳入了11名连续接受姑息性单层SDR手术的25岁以下儿童。所有患者的 MAS 评分均有所下降(平均 1.09 ± 0.66 分),且无手术发病率和死亡率发生。对于我们的系列病例的系统性回顾结果,除了4篇报告外,还纳入了274名患者。所有研究均显示,根据 MAS 评分,痉挛程度有所减轻(平均范围为 1.09-3.2 分)。此外,在两项研究中,上肢痉挛也显示出 MAS 评分的降低(范围为 1.7-2.8 分)。72%的患者 GMFM-66 评分有所改善,78%的患者膀胱功能有所改善。根据PROMs,92%的患者/护理人员对手术后的效果和生活质量表示满意。发生了两次伤口感染(2.7%)和一次脑脊液渗漏(1.3%),但没有出现与手术相关的死亡病例:这项分析表明,CP 和 GMFCS IV 级和 V 级患者的痉挛、日常护理和舒适度均有所改善。目前仍需要根据 MAS、GMFM-66 和 PROMs 对姑息性单级 SDR 的疗效进行更大规模的队列分析,这也是未来研究的重点。系统综述注册编号:CRD42024495762 (https://www.crd.york.ac.uk/prospero/)。
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引用次数: 0
Neurosurgical treatment of spasticity: a potential return to the cerebellum. 痉挛的神经外科治疗:小脑的潜在回归。
IF 3.3 2区 医学 Q2 CLINICAL NEUROLOGY Pub Date : 2024-06-01 DOI: 10.3171/2024.3.FOCUS2446
Daniel D Cummins, Hyun Joo Park, Fedor Panov

Objective: Neurosurgical targeting of the cerebellar dentate nucleus via ablative dentatotomy and stimulation of the dentate nucleus was historically used for effective treatment of spasticity. Yet for decades, neurosurgical treatment of spasticity targeting the cerebellum was bypassed in favor of alternative treatments such as intrathecal baclofen pumps and selective dorsal rhizotomies. Cerebellar neuromodulation has recently reemerged as a promising and effective therapy for spasticity and related movement disorders.

Methods: In this narrative review, the authors contextualize the historical literature of cerebellar neuromodulation, comparing it with modern approaches and exploring future directions with regard to cerebellar neuromodulation for spasticity.

Results: Neurosurgical intervention on the cerebellum dates to the use of dentatotomy in the 1960s, which had progressed to electrical stimulation of the cerebellar cortex and dentate nucleus by the 1980s. By 2024, modern neurosurgical approaches such as tractography-based targeting of the dentate nucleus and transcranial magnetic stimulation of cerebellar cortex have demonstrated promise for treating spasticity.

Conclusions: Cerebellar neuromodulation of the dentate nucleus and cerebellar cortex are promising therapies for severe cases of spasticity. Open areas for exploration in the field include the following: tractography-based targeting, adaptive cerebellar stimulation, and investigations into the network dynamics between the cerebellar cortex, deep cerebellar nuclei, and the subcortical and cortical structures of the cerebrum.

目的:历史上,通过消融齿状核切除术和刺激齿状核的神经外科手术以小脑齿状核为目标可有效治疗痉挛。然而数十年来,针对小脑的痉挛神经外科治疗一直被绕过,转而采用鞘内巴氯芬泵和选择性背根切断术等替代治疗方法。最近,小脑神经调控再次成为治疗痉挛和相关运动障碍的一种前景广阔的有效疗法:在这篇叙述性综述中,作者对小脑神经调控的历史文献进行了梳理,将其与现代方法进行了比较,并探讨了小脑神经调控治疗痉挛的未来方向:结果:对小脑的神经外科干预可追溯到 20 世纪 60 年代的齿状核切除术,到 20 世纪 80 年代发展到对小脑皮质和齿状核进行电刺激。到2024年,基于束成像的齿状核靶向治疗和小脑皮质经颅磁刺激等现代神经外科方法已显示出治疗痉挛的前景:结论:齿状核和小脑皮质的小脑神经调节是治疗严重痉挛病例的有前途的疗法。该领域有待探索的领域包括:基于小脑束成像的靶向治疗、自适应小脑刺激,以及对小脑皮质、小脑深核、大脑皮质下和皮质结构之间网络动力学的研究。
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引用次数: 0
Letter to the Editor. Considerations for the value of extended reality versus ex cathedra format for neuroanatomy education. 致编辑的信。对神经解剖学教育中扩展现实与传授形式价值的思考。
IF 3.3 2区 医学 Q2 CLINICAL NEUROLOGY Pub Date : 2024-06-01 DOI: 10.3171/2024.2.FOCUS2485
Marios Salmas, Dimitrios Chytas, George Noussios, Theano Demesticha, Angelo V Vasiliadis, Theodore Troupis
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引用次数: 0
The burden of skull base chordomas: insights from a meta-analysis of observational studies 颅底脊索瘤的负担:观察性研究荟萃分析的启示
IF 4.1 2区 医学 Q2 CLINICAL NEUROLOGY Pub Date : 2024-05-01 DOI: 10.3171/2024.3.focus23922
Giorgio Fiore, Edoardo Porto, Giulio A. Bertani, Hani J. Marcus, Andrea Saladino, Gustavo Pradilla, Francesco DiMeco, Marco Locatelli
OBJECTIVE

The aim of this study was to provide a quantitative synthesis of the survival outcomes for patients with skull base chordomas, focusing on the role of 1) the extent of resection (gross-total [GTR] vs non-GTR), 2) the type of surgery (primary vs revision), 3) tumor histology, and 4) the different use of adjuvant therapies (proton beam radiotherapy [PBRT], photon radiotherapy [RT], or none).

METHODS

A systematic review with a meta-analysis was conducted following the 2020 PRISMA guidelines. Observational studies describing adult and pediatric patient cohorts harboring skull base chordomas were included. The primary outcome measures were represented by the 5-year overall survival (OS) and progression-free survival (PFS) rates. The main intervention effects were represented by the extent of resection (GTR vs non-GTR), type of surgical excision (primary vs revision surgeries), tumor histology, and the different use of adjuvant therapies (PBRT, RT, or none). The pooled estimates were calculated using random forest models. The risk of bias was evaluated using the Joanna Briggs Institute checklist for case series.

RESULTS

Six hundred forty-four studies were identified through a database and register search. After study selection, 51 studies and 3871 patients were included in the meta-analysis. The overall 5-year OS rate was 73%, which increased to 84% among patients undergoing GTR. The overall 5-year PFS rate was 52%, increasing to 74% for patients receiving GTR. The 5-year OS and PFS rates for patients undergoing PBRT were 86% and 71%, compared with 71% and 54% for patients receiving RT, and 55% and 25% when no adjuvant treatments were used. Patients undergoing their first surgery had 2.13-fold greater chances of being disease-free and 1.4-fold greater chances of being alive at 5 years follow-up compared with patients who received a revision surgery. Patients harboring chondroid chordomas had 1.13- and 1.9-fold greater chances of being alive at 5 years compared with patients with conventional and de-differentiated chordomas, respectively. The overall risk of bias was low in the included studies.

CONCLUSIONS

The results of this comprehensive meta-analysis highlight the tremendous impact of GTR and adjuvant PBRT on improving OS and PFS of patients harboring skull base chordomas, with better survival rates demonstrated for patients with chondroid tumors. Even in experienced hands, the rate of surgical morbidity remains high. Proper management in high-volume centers is mandatory to reach the expected resection goal at the first surgical attempt and to reduce surgical morbidity. The introduction of the endoscopic endonasal approach was related to improved surgical and functional outcomes.

目的本研究旨在对颅底脊索瘤患者的生存结果进行定量综述,重点关注以下因素的作用:1)切除范围(全切除 [GTR] 与非全切除 [GTR]);2)手术类型(原发性与翻修性);3)肿瘤组织学;4)辅助疗法的不同使用(质子束放疗 [PBRT]、光子放疗 [RT] 或不使用)。方法按照 2020 年 PRISMA 指南进行了系统综述和荟萃分析。研究纳入了描述颅底脊索瘤成人和儿童患者队列的观察性研究。主要结局指标以 5 年总生存率(OS)和无进展生存率(PFS)表示。主要干预效应由切除范围(GTR 与非 GTR)、手术切除类型(初次手术与翻修手术)、肿瘤组织学以及辅助疗法的不同使用(PBRT、RT 或无)来表示。使用随机森林模型计算了汇总的估计值。采用乔安娜-布里格斯研究所(Joanna Briggs Institute)的病例系列检查表对偏倚风险进行了评估。经过研究筛选,51项研究和3871名患者被纳入荟萃分析。总体5年OS率为73%,在接受GTR治疗的患者中,OS率增至84%。总体5年PFS率为52%,接受GTR治疗的患者PFS率增至74%。接受PBRT治疗的患者的5年OS率和PFS率分别为86%和71%,而接受RT治疗的患者的5年OS率和PFS率分别为71%和54%,未接受辅助治疗的患者的5年OS率和PFS率分别为55%和25%。与接受复查手术的患者相比,接受首次手术的患者在随访5年后无病生存的几率要高出2.13倍,生存几率要高出1.4倍。与传统脊索瘤和去分化脊索瘤患者相比,患有软骨性脊索瘤的患者5年后存活的几率分别高出1.13倍和1.9倍。结论这项综合荟萃分析的结果凸显了GTR和辅助PBRT对改善颅底脊索瘤患者OS和PFS的巨大影响,软骨肿瘤患者的生存率更高。即使是经验丰富的医生,手术发病率仍然很高。要想在首次手术中达到预期的切除目标,并降低手术发病率,就必须在手术量大的中心进行适当的管理。内窥镜鼻内入路的引入改善了手术和功能效果。
{"title":"The burden of skull base chordomas: insights from a meta-analysis of observational studies","authors":"Giorgio Fiore, Edoardo Porto, Giulio A. Bertani, Hani J. Marcus, Andrea Saladino, Gustavo Pradilla, Francesco DiMeco, Marco Locatelli","doi":"10.3171/2024.3.focus23922","DOIUrl":"https://doi.org/10.3171/2024.3.focus23922","url":null,"abstract":"OBJECTIVE\u0000<p>The aim of this study was to provide a quantitative synthesis of the survival outcomes for patients with skull base chordomas, focusing on the role of 1) the extent of resection (gross-total [GTR] vs non-GTR), 2) the type of surgery (primary vs revision), 3) tumor histology, and 4) the different use of adjuvant therapies (proton beam radiotherapy [PBRT], photon radiotherapy [RT], or none).</p>METHODS\u0000<p>A systematic review with a meta-analysis was conducted following the 2020 PRISMA guidelines. Observational studies describing adult and pediatric patient cohorts harboring skull base chordomas were included. The primary outcome measures were represented by the 5-year overall survival (OS) and progression-free survival (PFS) rates. The main intervention effects were represented by the extent of resection (GTR vs non-GTR), type of surgical excision (primary vs revision surgeries), tumor histology, and the different use of adjuvant therapies (PBRT, RT, or none). The pooled estimates were calculated using random forest models. The risk of bias was evaluated using the Joanna Briggs Institute checklist for case series.</p>RESULTS\u0000<p>Six hundred forty-four studies were identified through a database and register search. After study selection, 51 studies and 3871 patients were included in the meta-analysis. The overall 5-year OS rate was 73%, which increased to 84% among patients undergoing GTR. The overall 5-year PFS rate was 52%, increasing to 74% for patients receiving GTR. The 5-year OS and PFS rates for patients undergoing PBRT were 86% and 71%, compared with 71% and 54% for patients receiving RT, and 55% and 25% when no adjuvant treatments were used. Patients undergoing their first surgery had 2.13-fold greater chances of being disease-free and 1.4-fold greater chances of being alive at 5 years follow-up compared with patients who received a revision surgery. Patients harboring chondroid chordomas had 1.13- and 1.9-fold greater chances of being alive at 5 years compared with patients with conventional and de-differentiated chordomas, respectively. The overall risk of bias was low in the included studies.</p>CONCLUSIONS\u0000<p>The results of this comprehensive meta-analysis highlight the tremendous impact of GTR and adjuvant PBRT on improving OS and PFS of patients harboring skull base chordomas, with better survival rates demonstrated for patients with chondroid tumors. Even in experienced hands, the rate of surgical morbidity remains high. Proper management in high-volume centers is mandatory to reach the expected resection goal at the first surgical attempt and to reduce surgical morbidity. The introduction of the endoscopic endonasal approach was related to improved surgical and functional outcomes.</p>","PeriodicalId":19187,"journal":{"name":"Neurosurgical focus","volume":"155 1","pages":""},"PeriodicalIF":4.1,"publicationDate":"2024-05-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"140835267","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":2,"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
Stereotactic radiosurgery in the management of skull base chordomas: a comprehensive systematic review and meta-analysis 立体定向放射外科治疗颅底脊索瘤:全面系统回顾和荟萃分析
IF 4.1 2区 医学 Q2 CLINICAL NEUROLOGY Pub Date : 2024-05-01 DOI: 10.3171/2024.2.focus249
Seyed Farzad Maroufi, Mohammad Sadegh Fallahi, Mohammadmahdi Sabahi, Seyede Parmis Maroufi, Jason P. Sheehan
OBJECTIVE

Chordoma is a primary bone tumor with limited literature on its management because of its rarity. Resection, while considered the first-line treatment, does not always provide adequate tumor control. In this systematic review, the authors aimed to provide comprehensive insights by managing these tumors with stereotactic radiosurgery (SRS).

METHODS

A systematic review was conducted according to PRISMA guidelines using the PubMed, Scopus, Web of Science, Embase, and Cochrane Library databases. Search terms included chordoma and radiosurgery and their equivalent terms. Data on baseline characteristics, SRS details, and outcomes were extracted. The Joanna Briggs Institute checklist was used to assess risk of bias. A meta-analysis was performed on relevant variables.

RESULTS

A total of 33 eligible studies encompassing 714 patients with skull base chordomas were included. Most studies had a low risk of bias. Patients, predominantly male (57.37%) with a mean age of 46.54 years, exhibited a conventional chordoma subtype (74.77%) and primary lesions (77.91%), mainly in the clivus (98.04%). The mean lesion volume was 13.49 cm3, and 96.68% of patients had undergone prior surgical attempts. Gamma Knife radiosurgery (88.76%) was the predominant SRS method. Radiologically, 27.19% of patients experienced tumor regression, while 55.02% showed no signs of disease progression at the latest follow-up. Progression occurred after a mean of 48.02 months. Symptom improvement was noted in 27.98% of patients. Radiosurgery was associated with a relatively low overall adverse event rate (11.94%), mainly cranial nerve deficits (8.72%). Meta-regression revealed that age and primary lesion type influenced symptom improvement, while factors like extent of resection, radiotherapy, and SRS type affected adverse event rates.

CONCLUSIONS

This systematic review provides evidence on the safety and effectiveness of radiosurgery in the management of skull base chordomas. Local tumor control was achieved in the majority of patients treated with SRS. Various baseline characteristics and SRS features have been analyzed to identify modifying factors for each outcome to provide a framework for informed decision-making when managing these patients.

目的脊索瘤是一种原发性骨肿瘤,由于其罕见性,有关其治疗的文献十分有限。切除术虽然被认为是一线治疗方法,但并不总能充分控制肿瘤。在这篇系统性综述中,作者旨在通过立体定向放射外科(SRS)治疗这些肿瘤提供全面的见解。方法根据 PRISMA 指南,使用 PubMed、Scopus、Web of Science、Embase 和 Cochrane Library 数据库进行了系统性综述。检索词包括脊索瘤和放射外科及其等效词。提取了有关基线特征、SRS细节和结果的数据。采用乔安娜-布里格斯研究所(Joanna Briggs Institute)核对表评估偏倚风险。结果共纳入 33 项符合条件的研究,包括 714 名颅底脊索瘤患者。大多数研究的偏倚风险较低。患者主要为男性(57.37%),平均年龄为46.54岁,表现为传统脊索瘤亚型(74.77%)和原发病灶(77.91%),主要位于颅骨(98.04%)。平均病变体积为13.49立方厘米,96.68%的患者曾接受过手术治疗。伽玛刀放射手术(88.76%)是最主要的 SRS 方法。从放射学角度看,27.19%的患者肿瘤消退,55.02%的患者在最近的随访中没有疾病进展的迹象。病情恶化的平均时间为 48.02 个月。27.98%的患者症状有所改善。放射手术的总体不良反应率相对较低(11.94%),主要是颅神经功能缺损(8.72%)。元回归显示,年龄和原发病灶类型影响症状的改善,而切除范围、放疗和 SRS 类型等因素则影响不良事件发生率。大多数接受SRS治疗的患者都实现了局部肿瘤控制。对各种基线特征和SRS特征进行了分析,以确定每种结果的影响因素,从而为管理这些患者时的知情决策提供一个框架。
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引用次数: 0
The utility of inflammatory biomarkers in predicting overall survival and recurrence in skull base chordoma 炎症生物标志物在预测颅底脊索瘤总体生存率和复发率方面的作用
IF 4.1 2区 医学 Q2 CLINICAL NEUROLOGY Pub Date : 2024-05-01 DOI: 10.3171/2024.2.focus2421
Meagan Hoppe, Zachary C. Gersey, Nallammai Muthiah, Hussein M. Abdallah, Tritan Plute, Hussam Abou-Al-Shaar, Eric W. Wang, Carl H. Snyderman, Georgios A. Zenonos, Paul A. Gardner
OBJECTIVE

Numerous studies have investigated the impact of inflammatory factors in cancer, yet few attempts have been made to investigate these markers in skull base chordoma (SBC). Inflammatory values including neutrophil-lymphocyte ratio (NLR), platelet-lymphocyte ratio (PLR), lymphocyte-monocyte ratio (LMR), systemic immune inflammation index (SII), and systemic inflammation response index (SIRI) can serve as prognostic markers in various cancers. This study aimed to determine whether these inflammatory factors influence overall survival (OS) or progression-free survival (PFS) in patients with primary SBC.

METHODS

The electronic medical records of patients with primary SBC who underwent resection from 2001 to 2020 were retrospectively reviewed for the associations of sex, age at diagnosis, preoperative steroid use, tumor volume, extent of resection, adjuvant radiation after surgery, tumor metastasis, Ki-67 index, percent homozygous deletion of 9p23 and percent 1p36 loss, and potential prognostic inflammatory markers of NLR, PLR, LMR, SII, and SIRI with the primary outcome measures of OS and PFS. Maximum log-rank statistical tests were used to determine inflammatory marker thresholds for grouping prior to Kaplan-Meier and Cox proportional hazards analysis for OS and PFS of the elucidated groups.

RESULTS

The cohort included 115 primary SBC patients. The mean ± SD tumor volume was 23.0 ± 28.0 cm3, 73% of patients received gross-total resection, 40% received postoperative radiation, 25% had local recurrence, and 6% had subsequent metastatic disease (mean follow-up 47.2 months). Univariable Cox analysis revealed that NLR (p < 0.01), PLR (p = 0.04), LMR (p = 0.04), SII (p < 0.01), and SIRI (p < 0.01) were independently associated with PFS. Additionally, NLR (p = 0.05) and SII (p = 0.03) were significant in multivariable Cox analysis of PFS. However, both univariable and multivariable Cox analysis revealed no correlations with OS.

CONCLUSIONS

The routine assessment of inflammatory biomarkers such as NLR and SIRI could have prognostic value in postresection SBC patients.

目的:已有大量研究探讨了炎症因素对癌症的影响,但很少有人尝试对颅底脊索瘤(SBC)中的这些标记物进行研究。包括中性粒细胞-淋巴细胞比值(NLR)、血小板-淋巴细胞比值(PLR)、淋巴细胞-单核细胞比值(LMR)、全身免疫炎症指数(SII)和全身炎症反应指数(SIRI)在内的炎症值可作为各种癌症的预后指标。本研究旨在确定这些炎症因子是否会影响原发性SBC患者的总生存期(OS)或无进展生存期(PFS)。方法回顾性分析2001年至2020年期间接受切除术的原发性SBC患者的电子病历,以了解性别、诊断年龄、术前类固醇使用情况、肿瘤体积、切除范围、术后辅助放疗、肿瘤转移、Ki-67指数、9p23同源缺失百分比和1p36缺失百分比以及NLR、PLR、LMR、SII和SIRI等潜在预后炎症标志物与OS和PFS主要结局指标的相关性。在对阐明组的 OS 和 PFS 进行 Kaplan-Meier 和 Cox 比例危险度分析之前,使用最大对数秩统计检验确定炎症标志物分组阈值。73%的患者接受了大体全切除术,40%接受了术后放疗,25%出现局部复发,6%出现后续转移性疾病(平均随访47.2个月)。单变量 Cox 分析显示,NLR(p < 0.01)、PLR(p = 0.04)、LMR(p = 0.04)、SII(p < 0.01)和 SIRI(p < 0.01)与 PFS 独立相关。此外,NLR(p = 0.05)和 SII(p = 0.03)在 PFS 的多变量 Cox 分析中具有显著性。结论常规评估 NLR 和 SIRI 等炎症生物标志物可能对 SBC 切除术后患者有预后价值。
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引用次数: 0
Introduction. Chordoma: updates and advances. 导言。脊索瘤:最新进展。
IF 3.3 2区 医学 Q2 CLINICAL NEUROLOGY Pub Date : 2024-05-01 DOI: 10.3171/2024.3.FOCUS23735
Paul A Gardner, Sebastien Froelich, Ziya L Gokaslan, Shannon M MacDonald, Maria Peris Celda, Shaan M Raza, Georgios A Zenonos
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引用次数: 0
Proton versus photon adjuvant radiotherapy: a multicenter comparative evaluation of recurrence following spinal chordoma resection 质子与光子辅助放疗:脊索瘤切除术后复发情况的多中心比较评估
IF 4.1 2区 医学 Q2 CLINICAL NEUROLOGY Pub Date : 2024-05-01 DOI: 10.3171/2024.2.focus23927
Abdul Karim Ghaith, Ryan Nguyen, Victor Gabriel El-Hajj, Alaa Montaser, Gaetano De Biase, Krishnan Ravindran, Carlos Perez-Vega, Seung Jin Lee, Asmina Dominari, Umberto Battistin, Paola Suárez-Meade, Charbel Moussalem, Naresh P. Patel, Maziyar A. Kalani, Michelle J. Clarke, Peter S. Rose, Mohamad Bydon, Kingsley Abode-Iyamah, Oluwaseun O. Akinduro
OBJECTIVE

Chordomas are rare tumors of the skull base and spine believed to arise from the vestiges of the embryonic notochord. These tumors are locally aggressive and frequently recur following resection and adjuvant radiotherapy. Proton therapy has been introduced as a tissue-sparing option because of the higher level of precision that proton-beam techniques offer compared with traditional photon radiotherapy. This study aimed to compare recurrence in patients with chordomas receiving proton versus photon radiotherapy following resection by applying tree-based machine learning models.

METHODS

The clinical records of all patients treated with resection followed by adjuvant proton or photon radiotherapy for chordoma at Mayo Clinic were reviewed. Patient demographics, type of surgery and radiotherapy, tumor recurrence, and other variables were extracted. Decision tree classifiers were trained and tested to predict long-term recurrence based on unseen data using an 80/20 split.

RESULTS

Fifty-three patients with a mean ± SD age of 55.2 ± 13.4 years receiving surgery and adjuvant proton or photon therapy to treat chordoma were identified; most patients were male. Gross-total resection was achieved in 54.7% of cases. Proton therapy was the most common adjuvant radiotherapy (84.9%), followed by conventional or external-beam radiation therapy (9.4%) and stereotactic radiosurgery (5.7%). Patients receiving proton therapy exhibited a 40% likelihood of having recurrence, significantly lower than the 88% likelihood observed in those treated with nonproton therapy. This was confirmed on logistic regression analysis adjusted for extent of tumor resection and tumor location, which revealed that proton adjuvant radiotherapy was associated with a decreased risk of recurrence (OR 0.1, 95% CI 0.01–0.71; p = 0.047) compared with photon therapy. The decision tree algorithm predicted recurrence with an accuracy of 90% (95% CI 55.5%–99.8%), with the lowest risk of recurrence observed in patients receiving gross-total resection with adjuvant proton therapy (23%).

CONCLUSIONS

Following resection, adjuvant proton therapy was associated with a lower risk of chordoma recurrence compared with photon therapy. The described machine learning models were able to predict tumor progression based on the extent of tumor resection and adjuvant radiotherapy modality used.

目的脊索瘤是一种罕见的颅底和脊柱肿瘤,据信是由胚胎脊索的残余部分产生的。这些肿瘤具有局部侵袭性,切除和辅助放疗后经常复发。与传统的光子放疗相比,质子束技术具有更高的精确度,因此质子疗法被作为一种保护组织的选择而引入。本研究旨在通过应用基于树的机器学习模型,比较脊索瘤患者切除术后接受质子放疗与光子放疗的复发率。方法回顾梅奥诊所所有脊索瘤患者切除术后辅助质子或光子放疗的临床记录。提取了患者的人口统计学特征、手术和放疗类型、肿瘤复发以及其他变量。结果53名患者接受了手术和质子或光子辅助疗法治疗脊索瘤,平均(±SD)年龄为55.2±13.4岁;大多数患者为男性。54.7%的病例实现了大体全切除。质子疗法是最常见的辅助放射疗法(84.9%),其次是传统或体外放射疗法(9.4%)和立体定向放射手术(5.7%)。接受质子治疗的患者复发的可能性为40%,明显低于接受非质子治疗的患者88%的复发可能性。根据肿瘤切除范围和肿瘤位置调整后进行的逻辑回归分析证实了这一点,该分析表明,与光子疗法相比,质子辅助放疗降低了复发风险(OR 0.1,95% CI 0.01-0.71;P = 0.047)。决策树算法预测复发的准确率为90%(95% CI 55.5%-99.8%),其中接受全切除术并辅助质子治疗的患者复发风险最低(23%)。所述机器学习模型能够根据肿瘤切除范围和辅助放疗方式预测肿瘤进展。
{"title":"Proton versus photon adjuvant radiotherapy: a multicenter comparative evaluation of recurrence following spinal chordoma resection","authors":"Abdul Karim Ghaith, Ryan Nguyen, Victor Gabriel El-Hajj, Alaa Montaser, Gaetano De Biase, Krishnan Ravindran, Carlos Perez-Vega, Seung Jin Lee, Asmina Dominari, Umberto Battistin, Paola Suárez-Meade, Charbel Moussalem, Naresh P. Patel, Maziyar A. Kalani, Michelle J. Clarke, Peter S. Rose, Mohamad Bydon, Kingsley Abode-Iyamah, Oluwaseun O. Akinduro","doi":"10.3171/2024.2.focus23927","DOIUrl":"https://doi.org/10.3171/2024.2.focus23927","url":null,"abstract":"OBJECTIVE\u0000<p>Chordomas are rare tumors of the skull base and spine believed to arise from the vestiges of the embryonic notochord. These tumors are locally aggressive and frequently recur following resection and adjuvant radiotherapy. Proton therapy has been introduced as a tissue-sparing option because of the higher level of precision that proton-beam techniques offer compared with traditional photon radiotherapy. This study aimed to compare recurrence in patients with chordomas receiving proton versus photon radiotherapy following resection by applying tree-based machine learning models.</p>\u0000METHODS\u0000<p>The clinical records of all patients treated with resection followed by adjuvant proton or photon radiotherapy for chordoma at Mayo Clinic were reviewed. Patient demographics, type of surgery and radiotherapy, tumor recurrence, and other variables were extracted. Decision tree classifiers were trained and tested to predict long-term recurrence based on unseen data using an 80/20 split.</p>\u0000RESULTS\u0000<p>Fifty-three patients with a mean ± SD age of 55.2 ± 13.4 years receiving surgery and adjuvant proton or photon therapy to treat chordoma were identified; most patients were male. Gross-total resection was achieved in 54.7% of cases. Proton therapy was the most common adjuvant radiotherapy (84.9%), followed by conventional or external-beam radiation therapy (9.4%) and stereotactic radiosurgery (5.7%). Patients receiving proton therapy exhibited a 40% likelihood of having recurrence, significantly lower than the 88% likelihood observed in those treated with nonproton therapy. This was confirmed on logistic regression analysis adjusted for extent of tumor resection and tumor location, which revealed that proton adjuvant radiotherapy was associated with a decreased risk of recurrence (OR 0.1, 95% CI 0.01–0.71; p = 0.047) compared with photon therapy. The decision tree algorithm predicted recurrence with an accuracy of 90% (95% CI 55.5%–99.8%), with the lowest risk of recurrence observed in patients receiving gross-total resection with adjuvant proton therapy (23%).</p>\u0000CONCLUSIONS\u0000<p>Following resection, adjuvant proton therapy was associated with a lower risk of chordoma recurrence compared with photon therapy. The described machine learning models were able to predict tumor progression based on the extent of tumor resection and adjuvant radiotherapy modality used.</p>","PeriodicalId":19187,"journal":{"name":"Neurosurgical focus","volume":"60 1","pages":""},"PeriodicalIF":4.1,"publicationDate":"2024-05-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"140835628","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":2,"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
Long-term outcome of primary clival chordomas: a single-center retrospective study with an emphasis on the timing of recurrences based on the primary treatment 原发性蝶骨脊索瘤的长期预后:一项单中心回顾性研究,重点是根据初治方法确定复发时间
IF 4.1 2区 医学 Q2 CLINICAL NEUROLOGY Pub Date : 2024-05-01 DOI: 10.3171/2024.2.focus23924
Sukwoo Hong, Yuki Shinya, Anita Mahajan, Nadia N. Laack, Erin K. O’Brien, Janalee K. Stokken, Jeffrey R. Janus, Aditya Raghunathan, Michael J. Link, Jamie J. Van Gompel
OBJECTIVE

This study aimed to provide data on extended outcomes in primary clival chordomas, focusing on progression-free survival (PFS) and overall survival (OS).

METHODS

A retrospective single-center analysis was conducted on patients with clival chordoma treated between 1987 and 2022 using surgery, stereotactic radiosurgery, or proton radiation therapy (PRT).

RESULTS

The study included 100 patients (median age 44 years, 51% male). Surgery was performed using the endoscopic endonasal approach in 71 patients (71%). Gross-total resection (GTR) or near-total resection (NTR) was attained in 39 patients (39%). Postoperatively, new cranial nerve deficits occurred in 7%, CSF leak in 4%, and meningitis in none of the patients. Radiation therapy was performed in 79 patients (79%), with PRT in 50 patients (50%) as the primary treatment. During the median follow-up period of 73 (interquartile range [IQR] 38–132) months, 41 recurrences (41%) and 31 deaths (31%) were confirmed. Patients with GTR/NTR had a median PFS of 41 (IQR 24–70) months. Patients with subtotal resection or biopsy had a median PFS of 38 (IQR 16–97) months. The median PFS of patients who received radiation therapy was 43 (IQR 26–86) months, while that of patients who did not receive radiation therapy was 18 (IQR 5–62) months. The Kaplan-Meier method showed that patients with GTR/NTR (p = 0.007) and those who received radiation therapy (p < 0.001) had longer PFS than their counterparts. The PFS rates following primary treatment at 5, 10, 15, and 20 years were 51%, 25%, 17%, and 7%, respectively. The OS rates at the same intervals were 84%, 60%, 42%, and 34%, respectively. Multivariate Cox regression analysis showed that age < 44 years (p = 0.02), greater extent of resection (EOR; p = 0.03), and radiation therapy (p < 0.001) were associated with lower recurrence rates. Another multivariate analysis showed that age < 44 years (p = 0.01), greater EOR (p = 0.04), and freedom from recurrence (p = 0.02) were associated with lower mortality rates. Regarding pathology data, brachyury was positive in 98%, pan-cytokeratin in 93%, epithelial membrane antigen in 85%, and S100 in 74%. No immunohistochemical markers were associated with recurrence.

CONCLUSIONS

In this study, younger age, maximal safe resection, and radiation therapy were important factors for longer PFS in patients with primary clival chordomas. Preventing recurrences played a crucial role in achieving longer OS.

方法对1987年至2022年间采用手术、立体定向放射外科或质子放疗(PRT)治疗的簇状脊索瘤患者进行了回顾性单中心分析。结果研究共纳入100名患者(中位年龄44岁,51%为男性)。71名患者(71%)采用内窥镜鼻内入路进行手术。39名患者(39%)实现了全切除(GTR)或近全切除(NTR)。术后,7%的患者出现新的颅神经功能缺损,4%的患者出现脑脊液漏,无一例患者出现脑膜炎。79名患者(79%)接受了放射治疗,50名患者(50%)接受了PRT作为主要治疗手段。中位随访期为 73 个月(四分位数间距 [IQR] 38-132),其中 41 例复发(41%),31 例死亡(31%)。GTR/NTR患者的中位生存期为41个月(IQR 24-70)。次全切除或活检患者的中位 PFS 为 38 个月(IQR 16-97)。接受放射治疗的患者的中位生存期为43(IQR 26-86)个月,而未接受放射治疗的患者的中位生存期为18(IQR 5-62)个月。卡普兰-梅耶法显示,GTR/NTR 患者(p = 0.007)和接受放疗的患者(p < 0.001)的 PFS 比同类患者长。初治后5年、10年、15年和20年的PFS率分别为51%、25%、17%和7%。相同时间间隔内的 OS 率分别为 84%、60%、42% 和 34%。多变量 Cox 回归分析显示,年龄 44 岁(p = 0.02)、切除范围更大(EOR;p = 0.03)和放疗(p = 0.001)与较低的复发率相关。另一项多变量分析显示,年龄为 44 岁(p = 0.01)、切除范围更大(p = 0.04)和无复发(p = 0.02)与较低的死亡率有关。在病理数据方面,98%的患者胸腺嘧啶阳性,93%的患者泛角蛋白阳性,85%的患者上皮膜抗原阳性,74%的患者 S100 阳性。结论 在这项研究中,年龄较小、最大安全切除和放疗是原发性簇状脊索瘤患者延长生存期的重要因素。预防复发对延长OS至关重要。
{"title":"Long-term outcome of primary clival chordomas: a single-center retrospective study with an emphasis on the timing of recurrences based on the primary treatment","authors":"Sukwoo Hong, Yuki Shinya, Anita Mahajan, Nadia N. Laack, Erin K. O’Brien, Janalee K. Stokken, Jeffrey R. Janus, Aditya Raghunathan, Michael J. Link, Jamie J. Van Gompel","doi":"10.3171/2024.2.focus23924","DOIUrl":"https://doi.org/10.3171/2024.2.focus23924","url":null,"abstract":"OBJECTIVE\u0000<p>This study aimed to provide data on extended outcomes in primary clival chordomas, focusing on progression-free survival (PFS) and overall survival (OS).</p>\u0000METHODS\u0000<p>A retrospective single-center analysis was conducted on patients with clival chordoma treated between 1987 and 2022 using surgery, stereotactic radiosurgery, or proton radiation therapy (PRT).</p>\u0000RESULTS\u0000<p>The study included 100 patients (median age 44 years, 51% male). Surgery was performed using the endoscopic endonasal approach in 71 patients (71%). Gross-total resection (GTR) or near-total resection (NTR) was attained in 39 patients (39%). Postoperatively, new cranial nerve deficits occurred in 7%, CSF leak in 4%, and meningitis in none of the patients. Radiation therapy was performed in 79 patients (79%), with PRT in 50 patients (50%) as the primary treatment. During the median follow-up period of 73 (interquartile range [IQR] 38–132) months, 41 recurrences (41%) and 31 deaths (31%) were confirmed. Patients with GTR/NTR had a median PFS of 41 (IQR 24–70) months. Patients with subtotal resection or biopsy had a median PFS of 38 (IQR 16–97) months. The median PFS of patients who received radiation therapy was 43 (IQR 26–86) months, while that of patients who did not receive radiation therapy was 18 (IQR 5–62) months. The Kaplan-Meier method showed that patients with GTR/NTR (p = 0.007) and those who received radiation therapy (p &lt; 0.001) had longer PFS than their counterparts. The PFS rates following primary treatment at 5, 10, 15, and 20 years were 51%, 25%, 17%, and 7%, respectively. The OS rates at the same intervals were 84%, 60%, 42%, and 34%, respectively. Multivariate Cox regression analysis showed that age &lt; 44 years (p = 0.02), greater extent of resection (EOR; p = 0.03), and radiation therapy (p &lt; 0.001) were associated with lower recurrence rates. Another multivariate analysis showed that age &lt; 44 years (p = 0.01), greater EOR (p = 0.04), and freedom from recurrence (p = 0.02) were associated with lower mortality rates. Regarding pathology data, brachyury was positive in 98%, pan-cytokeratin in 93%, epithelial membrane antigen in 85%, and S100 in 74%. No immunohistochemical markers were associated with recurrence.</p>\u0000CONCLUSIONS\u0000<p>In this study, younger age, maximal safe resection, and radiation therapy were important factors for longer PFS in patients with primary clival chordomas. Preventing recurrences played a crucial role in achieving longer OS.</p>","PeriodicalId":19187,"journal":{"name":"Neurosurgical focus","volume":"2 1","pages":""},"PeriodicalIF":4.1,"publicationDate":"2024-05-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"140829207","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":2,"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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Neurosurgical focus
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