Journal of Neuro-Oncology最新文献

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Long-term impact of bevacizumab for the treatment of brain radiation necrosis.
IF 3.2 2区 医学
Journal of Neuro-Oncology Pub Date : 2025-03-12 DOI: 10.1007/s11060-025-04979-1
Hila Nobel, Jonathan Ofer, Sara Faye Borenstein, Dror Limon, Omer Gal, Yosef Laviv, Andrew A Kanner, Tali Siegal, Shlomit Yust-Katz, Alexandra Benouaich-Amiel
{"title":"Long-term impact of bevacizumab for the treatment of brain radiation necrosis.","authors":"Hila Nobel, Jonathan Ofer, Sara Faye Borenstein, Dror Limon, Omer Gal, Yosef Laviv, Andrew A Kanner, Tali Siegal, Shlomit Yust-Katz, Alexandra Benouaich-Amiel","doi":"10.1007/s11060-025-04979-1","DOIUrl":"https://doi.org/10.1007/s11060-025-04979-1","url":null,"abstract":"<p><strong>Purpose: </strong>To evaluate short and long-term efficacy of bevacizumab (Bev), for the treatment of radiation necrosis (RN) in patients with brain metastasis after stereotactic radiosurgery (SRS).</p><p><strong>Methods: </strong>The database of a tertiary medical center was reviewed for all adult patients treated by Bev (from January 2018 to January 2023) for RN after having received SRS for BM. Clinical and MRI data were systematically collected at baseline, immediately after the completion of Bev treatment, and at 6, 12, and, when available, 24 months post-treatment.</p><p><strong>Results: </strong>The cohort included 23 patients with a total of 31 RN lesions (defined as target lesion) which have been previously treated by SRS, either as single-session SRS (27/31) or as fractionated stereotactic radiotherapy (4/31). Median follow-up time was 15 months (range: 8-28.5). Immediately after completion of Bev, 15 patients (65.2%) exhibited a complete/partial response, 6 (26.1%) had stable disease, and 2 had progressive disease (8.7%). thirteen patients (56%) improved clinically. Greater than 50% reduction in volume was observed in 84% of target lesions. At 12 months, among the 13 patients still evaluable (9 other being deceased, 1 loss to follow up), three continued to improve, and four remained stable. Median volume of target lesion was then 1.4 cm<sup>3</sup> (range 0.7-2.9) demonstrating a reduction of 67.4% compared to the initial target volume, which was 4.35 cm<sup>3</sup> (range 2.14-10.37). During the entire follow-up period, 11 patients experienced regrowth of the target lesion; median time to progression was 7 months. Five underwent Bev re-challenge, but only 2 responded.</p><p><strong>Conclusion: </strong>Bev for the treatment of SRS-induced RN was associated with a high initial response rate, significant lesion reduction, and prolonged clinical improvement. However, the high rate of lesion regrowth (50%) and poor response to Bev re-challenge highlight the complexity of diagnosis and treatment of RN.</p>","PeriodicalId":16425,"journal":{"name":"Journal of Neuro-Oncology","volume":" ","pages":""},"PeriodicalIF":3.2,"publicationDate":"2025-03-12","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143615761","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
Gamma Knife Radiosurgery for optic nerve sheath meningioma: comparison of efficacy and costs with radiotherapy under Korean health insurance system.
IF 3.2 2区 医学
Journal of Neuro-Oncology Pub Date : 2025-03-11 DOI: 10.1007/s11060-025-04986-2
Shang-Te Ma, Yong Chan Ahn, Do Hoon Lim, Jung-Il Lee, Ho Jun Seol, Won-Jae Lee, Yong Seok Im, Kyung-Ah Park, Yoon-Duck Kim, Kyung In Woo
{"title":"Gamma Knife Radiosurgery for optic nerve sheath meningioma: comparison of efficacy and costs with radiotherapy under Korean health insurance system.","authors":"Shang-Te Ma, Yong Chan Ahn, Do Hoon Lim, Jung-Il Lee, Ho Jun Seol, Won-Jae Lee, Yong Seok Im, Kyung-Ah Park, Yoon-Duck Kim, Kyung In Woo","doi":"10.1007/s11060-025-04986-2","DOIUrl":"https://doi.org/10.1007/s11060-025-04986-2","url":null,"abstract":"<p><strong>Purpose: </strong>To survey the real-world effectiveness and cost of optic nerve sheath meningioma (ONSM) treating with Gamma Knife Radiosurgery (GKRS), and compare with the external beam radiation therapy (EBRT).</p><p><strong>Methods: </strong>Retrospective, comparative study that included patients with primary ONSM treated with either GKRS or EBRT in Samsung Medical Center, Korea. The treatment response, and treatment costs were compared between GKRS and EBRT groups.</p><p><strong>Results: </strong>There were 34 adult patients with primary ONSM treated with either GKRS (n = 25) or EBRT (n = 9) (follow-up period: 6-207 months). The local tumor control rates (GKRS: 92%; EBRT: 100%; P = 1) and vision preservation rates (GKRS: 64%; EBRT: 67%; P = 1) were similar in both groups. The mean gross tumor volume (GTV) decreased by 21.4 ± 19.7% after GKRS and 26.4 ± 18.7% after EBRT (P = 0.4803). The complication rates did not differ between two modalities. Factors associated with better visual outcomes were pretreatment BCVA > 20/50 (odds ratio: 6.000, P = 0.0234) and the absence of intracranial tumor extension (odds ratio: 30.00, P = 0.0001). GKRS reduced the total costs of care by 43% under Korean National Health Insurance System (NHIS).</p><p><strong>Conclusion: </strong>This study revealed that GKRS and EBRT had similar treatment efficacy and safety profile in treating ONSMs. Instant management is advantageous when the BCVA deteriorates to 20/50 and tumors involve intracranially. Under the framework of Korean NHIS, GKRS contributed to less indirect cost estimates and was preferred by patients under shared decision-making process.</p>","PeriodicalId":16425,"journal":{"name":"Journal of Neuro-Oncology","volume":" ","pages":""},"PeriodicalIF":3.2,"publicationDate":"2025-03-11","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143605208","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
The Mickey Mouse's hand sign in brain MRI points out multinodular and vacuolating neuronal tumors in mesial temporal lobe structures.
IF 3.2 2区 医学
Journal of Neuro-Oncology Pub Date : 2025-03-11 DOI: 10.1007/s11060-025-04962-w
Valerio Frazzini, Bertrand Mathon, Natalia Shor, Lucia Nichelli, Franck Bielle, Vincent Navarro
{"title":"The Mickey Mouse's hand sign in brain MRI points out multinodular and vacuolating neuronal tumors in mesial temporal lobe structures.","authors":"Valerio Frazzini, Bertrand Mathon, Natalia Shor, Lucia Nichelli, Franck Bielle, Vincent Navarro","doi":"10.1007/s11060-025-04962-w","DOIUrl":"https://doi.org/10.1007/s11060-025-04962-w","url":null,"abstract":"<p><strong>Purpose: </strong>Multinodular and vacuolating neuronal tumor (MVNT) is a rarely diagnosed neoplastic lesion often associated with adult-onset focal seizures. In some situations, atypical MRI features of MVNT may mimic other long-term epilepsy associated tumors (LEATs) or diffuse low-grade gliomas. In such a context, the identification of distinct clinical markers is recommended.</p><p><strong>Methods: </strong>We retrospectively analyzed the neuroimaging features of histologically confirmed MVNT lesions of our epilepsy surgery cohort. All the identified MVNT patients had a mesial temporal lobe location. The MRI features from the MVNT population were compared to those from non-MVNT lesions randomly selected in our cohort. Non-MVNT lesions consisted of (i) other tumoral lesions belonging to LEATs group, (ii) infiltrative tumoral lesions such as grade 2, IDH-mutant Gliomas, and (iii) non-tumoral, highly epileptogenic lesions, such as hippocampal sclerosis.</p><p><strong>Results: </strong>MRI analysis of patients with MVNT noticed an unusual FLAIR hyperintense signal extending from the uncus to the anterior white commissure through the sublenticular region. This neuroimaging abnormality visually mimicked \"Mickey Mouse's hand\" on coronal plane. The \"Mickey Mouse's hand\" sign was identified in 6 patients out of 9 (66.7%) MVNT patients. We did not identify the Mickey Mouse's hand sign in other control lesions (0/11 ganglioglioma, 0/11 glioma, 0/150 hippocampal sclerosis), except in one patient with dysembryoplastic neuroepithelial tumors (DNET) (1/15; 6.7%). All patients showing the \"Mickey Mouse's hand\" sign were seizure-free after surgery.</p><p><strong>Conclusions: </strong>The Mickey Mouse's hand sign represents a straightforward and easy-to-remember neuroimaging feature, that does not require advanced MRI sequences and that could help clinicians facing mesial temporal lesions to rapidly raise the suspicion of MVNT diagnosis.</p>","PeriodicalId":16425,"journal":{"name":"Journal of Neuro-Oncology","volume":" ","pages":""},"PeriodicalIF":3.2,"publicationDate":"2025-03-11","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143597236","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
Impact of alternating electric fields therapy for newly diagnosed WHO grade 4 astrocytoma on patient survival: a real-world propensity-score adjusted prospective multicenter study.
IF 3.2 2区 医学
Journal of Neuro-Oncology Pub Date : 2025-03-11 DOI: 10.1007/s11060-025-04985-3
Peter Y M Woo, Jenny K S Pu, Lai-Fung Li, Desiree K K Wong, Victor K H Hui, Danny T M Chan, Michael W Y Lee, Tony K T Chan, Jason M K Ho, Ka-Man Cheung, Teresa P K Tse, Sarah S N Lau, Joyce S W Chow, Natalie M W Ko, Herbert H F Loong, Aya El-Helali, Tai-Chung Lam, Fung-Ching Cheung, Wai-Sang Poon
{"title":"Impact of alternating electric fields therapy for newly diagnosed WHO grade 4 astrocytoma on patient survival: a real-world propensity-score adjusted prospective multicenter study.","authors":"Peter Y M Woo, Jenny K S Pu, Lai-Fung Li, Desiree K K Wong, Victor K H Hui, Danny T M Chan, Michael W Y Lee, Tony K T Chan, Jason M K Ho, Ka-Man Cheung, Teresa P K Tse, Sarah S N Lau, Joyce S W Chow, Natalie M W Ko, Herbert H F Loong, Aya El-Helali, Tai-Chung Lam, Fung-Ching Cheung, Wai-Sang Poon","doi":"10.1007/s11060-025-04985-3","DOIUrl":"https://doi.org/10.1007/s11060-025-04985-3","url":null,"abstract":"<p><strong>Purpose: </strong>Alternating electric fields (AEF) therapy in addition to temozolomide chemoradiotherapy (TMZ CRT) is increasingly being recommended as first-line treatment for patients with newly-diagnosed WHO grade 4 astrocytoma. However, few have validated this treatment with real-world evidence.</p><p><strong>Methods: </strong>Consecutive adult patients with newly-diagnosed WHO grade 4 astrocytoma treated with adjuvant TMZ CRT across all neuro-oncology centers in Hong Kong were reviewed. Identified from a territory-wide prospective glioma registry, propensity-score matching (1:2) was performed to match patients that either received TMZ CRT with AEF or TMZ CRT alone. Matching was according to age, Karnofsky performance status, IDH-1 mutation, pMGMT methylation and extent of resection. The primary endpoint was overall survival (OS). Secondary endpoints were the incidence of AEF-associated adverse effects and mean monthly treatment compliance.</p><p><strong>Results: </strong>141 patients were reviewed, of whom 47 patients received AEF with TMZ CRT and 94 had CRT alone. Multivariate Cox proportional hazards analysis revealed that patients with pMGMT-methylated tumors (mOS: 30.8 months vs. 16.7 months [95% CI: 1.9-4.7] and those that received AEF (mOS: 22.8 vs. 14.3 months [95% CI: 1.9-4.7]) had longer OS. AEF therapy patients had a mOS benefit of 8.5 months. The mean monthly treatment compliance was 74 ± 12%. A compliance threshold of 60% conferred a survival benefit of 4.1 months (mOS: 21.5 months vs. 17.4 months [95% CI: 0.10-0.96]). The only identified AEF-associated adverse reaction was scalp dermatitis that occured in 77% (36/47) of patients.</p><p><strong>Conclusion: </strong>This post-approval study offers real-world evidence in support of the use of AEF therapy as first-line treatment.</p>","PeriodicalId":16425,"journal":{"name":"Journal of Neuro-Oncology","volume":" ","pages":""},"PeriodicalIF":3.2,"publicationDate":"2025-03-11","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143605210","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
Bevacizumab in recurrent glioblastoma: does dose matter? Our monocentric and comparative experience.
IF 3.2 2区 医学
Journal of Neuro-Oncology Pub Date : 2025-03-10 DOI: 10.1007/s11060-025-04992-4
Giulia Cerretti, Alberto Bosio, Giovanni Librizzi, Giovanna Pintacuda, Mario Caccese, Alessandro Salvalaggio, Marco Zoccarato, Alessandro Parisi, Marta Padovan, Marta Maccari, Francesco Cavallin, Luisa Bellu, Francesco Pasqualetti, Tamara Ius, Luca Denaro, Francesco Volpin, Marina Coppola, Sara Lonardi, Giuseppe Lombardi
{"title":"Bevacizumab in recurrent glioblastoma: does dose matter? Our monocentric and comparative experience.","authors":"Giulia Cerretti, Alberto Bosio, Giovanni Librizzi, Giovanna Pintacuda, Mario Caccese, Alessandro Salvalaggio, Marco Zoccarato, Alessandro Parisi, Marta Padovan, Marta Maccari, Francesco Cavallin, Luisa Bellu, Francesco Pasqualetti, Tamara Ius, Luca Denaro, Francesco Volpin, Marina Coppola, Sara Lonardi, Giuseppe Lombardi","doi":"10.1007/s11060-025-04992-4","DOIUrl":"https://doi.org/10.1007/s11060-025-04992-4","url":null,"abstract":"<p><strong>Purpose: </strong>Bevacizumab is an anti-angiogenetic treatment that can be used in patients with recurrent glioblastoma, but there are limited and controversial data on the optimal dose and schedule, associated toxicities and survival benefits of different doses.</p><p><strong>Methods: </strong>A retrospective analysis of patients with recurrent IDHwt glioblastoma treated with bevacizumab at the Veneto Institute of Oncology was performed. Patients received bevacizumab in 2 different schedules (5 mg/kg or 10 mg/kg q2w), as monotherapy or in combination with chemotherapy.</p><p><strong>Results: </strong>81 patients were analyzed, 33 received bevacizumab 5 mg/Kg, 48 received bevacizumab 10 mg/Kg. Median PFS was 4 months in both patients treated with 5 mg/kg and those treated with 10 mg/kg (p-value=0.08), median OS was 5 months in patients treated with 5 mg/kg and 7 months in those treated with 10 mg/kg (p-value=0.10). There was no difference in the use of steroid therapy between the two groups. The incidence of adverse events was not statistically different.</p><p><strong>Conclusions: </strong>There was no statistically significant difference in survival, PFS, response, toxicity and steroid reduction between the two different doses. These results may support the use of lower doses of the drug with comparable benefit for patients and with additional advantage in terms of health care costs.</p>","PeriodicalId":16425,"journal":{"name":"Journal of Neuro-Oncology","volume":" ","pages":""},"PeriodicalIF":3.2,"publicationDate":"2025-03-10","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143597234","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
The spectrum of radiation therapy options for craniopharyngioma: a systematic review.
IF 3.2 2区 医学
Journal of Neuro-Oncology Pub Date : 2025-03-10 DOI: 10.1007/s11060-025-05001-4
Paul M Harary, Sanjeeth Rajaram, Yusuke S Hori, David J Park, Steven D Chang
{"title":"The spectrum of radiation therapy options for craniopharyngioma: a systematic review.","authors":"Paul M Harary, Sanjeeth Rajaram, Yusuke S Hori, David J Park, Steven D Chang","doi":"10.1007/s11060-025-05001-4","DOIUrl":"https://doi.org/10.1007/s11060-025-05001-4","url":null,"abstract":"<p><strong>Purpose: </strong>Craniopharyngiomas (CPs) are rare, slow-growing brain tumors which originate in the sellar region. CPs may present with symptoms secondary to compression of surrounding structures, particularly the pituitary gland, and surgical removal has traditionally been the mainstay of treatment. However, due to high recurrence rates for CPs, especially when gross total resection is not feasible, radiotherapy has played an increasingly significant role in their management. Here, we review radiation modalities, treatment settings, and future directions in the management of CP. In addition, we outline emerging therapeutic combinations involving targeted therapies.</p><p><strong>Methods: </strong>A systematic review was performed in accordance with Preferred Reporting Items for Systematic Reviews and Meta-Analyses guidelines. A search of the MEDLINE/PubMed, Embase, and Web of Science databases was used for initial identification of articles. Included studies were then grouped into the following treatment modalities: conventional radiotherapy (CRT), intensity-modulated radiotherapy (IMRT), proton therapy, fractionated stereotactic radiotherapy (FSRT) /stereotactic radiosurgery (SRS), and brachytherapy.</p><p><strong>Results: </strong>A total of 60 studies met inclusion criteria, comprising 3041 patients, with a median sample size of 33 (range: 10-242) and age ranging from 6 to 55. This review included 8, 15, 3, 12, and 29 reports corresponding to brachytherapy, CRT, IMRT, proton therapy, and FSRT/SRS, respectively. Proton therapy and FSRT had the highest median 5-year progression-free survival (PFS), with rates of 92% and 89%, respectively. IMRT and proton therapy were primarily investigated in pediatric patients (median ages of 8.2 and 10.3 years, respectively). By comparison, FSRT and SRS research has mainly been in adult cohorts.</p><p><strong>Conclusions: </strong>Precision radiotherapy appears to be associated with high rates of tumor control in CP, suggesting these approaches bear further investigation. Selection of the appropriate radiation modality from those reviewed likely depends on several patient-specific factors. Important considerations include tumor location and volume, patient age, prior treatments for CP, and patient preference.</p>","PeriodicalId":16425,"journal":{"name":"Journal of Neuro-Oncology","volume":" ","pages":""},"PeriodicalIF":3.2,"publicationDate":"2025-03-10","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143597237","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
Introducing the operative value index for glioma surgery: an integration of quality-adjusted life years with time-driven activity-based costing.
IF 3.2 2区 医学
Journal of Neuro-Oncology Pub Date : 2025-03-10 DOI: 10.1007/s11060-025-04997-z
Advith Sarikonda, Danyal Quraishi, D Mitchell Self, Ashmal Sami, Steven Glener, Matthews Lan, Sanyam Ratan, Anthony Yulin Chen, Antony Fuleihan, Pranav Jain, Ayra Khan, Justin Santos, Conor Dougherty, Emily L Isch, Nicholas Clark, James J Evans, Kevin D Judy, Christopher J Farrell, Ahilan Sivaganesan
{"title":"Introducing the operative value index for glioma surgery: an integration of quality-adjusted life years with time-driven activity-based costing.","authors":"Advith Sarikonda, Danyal Quraishi, D Mitchell Self, Ashmal Sami, Steven Glener, Matthews Lan, Sanyam Ratan, Anthony Yulin Chen, Antony Fuleihan, Pranav Jain, Ayra Khan, Justin Santos, Conor Dougherty, Emily L Isch, Nicholas Clark, James J Evans, Kevin D Judy, Christopher J Farrell, Ahilan Sivaganesan","doi":"10.1007/s11060-025-04997-z","DOIUrl":"https://doi.org/10.1007/s11060-025-04997-z","url":null,"abstract":"<p><strong>Background: </strong>Although many studies have examined outcomes after glioma surgery, few have explored the factors driving variation in the cost-effectiveness of surgical care. In this study, we integrate granular time-driven activity-based costing (TDABC) methodology with quality-adjusted life years (QALYs) to measure the true \"value\" (outcomes achieved per dollar spent) of glioma surgery.</p><p><strong>Methods: </strong>176 glioma surgeries performed at a single institution were reviewed. Process maps were designed to identify all resources utilized in the intraoperative episode. Costing software was developed to automate the extraction of this data from the electronic medical record (EMR). QALYs were calculated based on progression-free survival (PFS) and 6-month postoperative Karnofsky Performance Status (KPS) scores. The Operative Value Index (OVI) was defined as the QALYs achieved per $1,000 spent intraoperatively. Multivariable regression models were performed to examine factors driving variability in both costs and OVI.</p><p><strong>Results: </strong>The median total cost of surgery was $6,987, most of which was driven by the cost of supplies ($3,804, 53%) and personnel ($1,635, 23%). The median QALY was 0.96, PFS was 403 days (1.1 years), and the OVI was 0.14. Multivariable regression analysis revealed that awake surgery was associated with $2,540 of additional cost compared to surgery under general anesthesia, while World Health Organization Grade III (p < 0.001) and Grade IV (p < 0.001) gliomas were associated with significantly lower OVI.</p><p><strong>Conclusions: </strong>This study establishes a scalable, EMR-based framework for evaluating surgical value by integrating cost with outcomes. We show that awake surgery is associated with significantly higher total cost, and that increasing glioma disease severity is associated with worse outcomes achieved per dollar spent.</p>","PeriodicalId":16425,"journal":{"name":"Journal of Neuro-Oncology","volume":" ","pages":""},"PeriodicalIF":3.2,"publicationDate":"2025-03-10","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143597235","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
Gene therapy and genome-editing for schwannoma in NF2-related schwannomatosis: current understanding and future directions.
IF 3.2 2区 医学
Journal of Neuro-Oncology Pub Date : 2025-03-07 DOI: 10.1007/s11060-025-04995-1
Ryota Tamura, Masahiro Yo, Masahiro Toda
{"title":"Gene therapy and genome-editing for schwannoma in NF2-related schwannomatosis: current understanding and future directions.","authors":"Ryota Tamura, Masahiro Yo, Masahiro Toda","doi":"10.1007/s11060-025-04995-1","DOIUrl":"https://doi.org/10.1007/s11060-025-04995-1","url":null,"abstract":"<p><p>NF2-related schwannomatosis (NF2-SWN) is a rare genetic disorder characterized by bilateral vestibular schwannomas. NF2-SWN represents a difficult management problem with most patients facing substantial morbidity and reduced life expectancy. Gene therapy involves replacing a faulty gene or adding a new gene in an attempt to cure disease or improve the patient's condition. Several studies of gene therapy for NF2-SWN have utilized adeno-associated viral vector serotype-1 (AAV1) to deliver apoptosis-inducing enzyme, the pore-forming protein gasdermin-D, apoptosis-associated speck-like protein containing a caspase recruitment domain, and functional merlin causing schwannoma regression in a xenograft mouse model. These studies support the potential therapeutic efficacy of gene therapy against NF2-SWN. Currently, gene therapy approaches primarily include bystander-killing effect by inducing immune responses, gene replacement or augmentation therapy, and gene knockdown and replacement combination approach through genome-editing technology. Although these gene therapeutic strategies have shown potential in preclinical animal model studies, they still face many specific challenges apart from the traditional challenges in gene therapy, such as immunogenicity, delivery vector, manufacturing, and long-term effects of treatments. In this article, we discuss the current understanding and future directions of gene therapy and genome-editing for schwannoma in NF2-SWN.</p>","PeriodicalId":16425,"journal":{"name":"Journal of Neuro-Oncology","volume":" ","pages":""},"PeriodicalIF":3.2,"publicationDate":"2025-03-07","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143585971","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
Extra-central nervous system metastasis from high-grade glioma: a single-institution experience.
IF 3.2 2区 医学
Journal of Neuro-Oncology Pub Date : 2025-03-06 DOI: 10.1007/s11060-025-04977-3
Christina Abi Faraj, Ian E McCutcheon, Maria A Gubbiotti, Subha Perni, Maria K Gule-Monroe, Kadir Akdemir, Victoria E Clark, Evan D Bander, Monica E Loghin, Sujit S Prabhu, Frederick F Lang, Jeffrey S Weinberg
{"title":"Extra-central nervous system metastasis from high-grade glioma: a single-institution experience.","authors":"Christina Abi Faraj, Ian E McCutcheon, Maria A Gubbiotti, Subha Perni, Maria K Gule-Monroe, Kadir Akdemir, Victoria E Clark, Evan D Bander, Monica E Loghin, Sujit S Prabhu, Frederick F Lang, Jeffrey S Weinberg","doi":"10.1007/s11060-025-04977-3","DOIUrl":"https://doi.org/10.1007/s11060-025-04977-3","url":null,"abstract":"<p><strong>Purpose: </strong>Extra-central nervous system metastasis (ECM) from glioblastoma and other high-grade gliomas (HGGs) is exceedingly rare, likely due to central nervous system barriers and the short overall survival (OS) in HGG patients, limiting the timeframe for metastasis. Improved treatments have extended survival, potentially increasing ECM incidence, though mechanisms remain unclear.</p><p><strong>Methods: </strong>This retrospective study examines HGG patients (n = 16) with ECM treated at The University of Texas M. D. Anderson Cancer Center from 1993 to 2023.</p><p><strong>Results: </strong>Median age at HGG and ECM diagnoses were 33.6 and 35.1 years, respectively, with a slight female predominance. Diagnoses included glioblastoma, IDH-wildtype WHO Grade 4 (n = 11), epithelioid glioblastoma WHO Grade 4 (n = 2), astrocytoma IDH-mutant WHO Grade 4 (n = 2), and H3K27-altered diffuse midline glioma (n = 1). Median interval from HGG to ECM diagnosis was 10 months. The temporal lobe was the most common HGG site, with ECM primarily in cervical lymph nodes, bone, parotid gland, and cranial soft tissues. Genomic profiling identified TP53, EGFR, RB1, NF1, TERT promoter, and BRAF V600E mutations. Median OS from HGG diagnosis was 23.4 months, and median OS following ECM diagnosis was 5.9 months. Chemotherapy and radiotherapy to ECM sites extended survival. Leptomeningeal disease was present in 50% of cases and correlated with worse prognosis. ECM typically developed in advanced disease stages.</p><p><strong>Conclusion: </strong>This study highlights genomic alterations, management, and outcomes associated with ECM in HGG. Tumor spread may stem from neurosurgical manipulation and occur via hematogenous and/or lymphatic routes. Multimodal treatment extends survival. Targeted therapies based on molecular profiles should be explored.</p>","PeriodicalId":16425,"journal":{"name":"Journal of Neuro-Oncology","volume":" ","pages":""},"PeriodicalIF":3.2,"publicationDate":"2025-03-06","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143585967","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
Peritumoral edema resolves infrequently in surgically treated patients with intracranial meningioma- a retrospective study of 279 meningioma patients.
IF 3.2 2区 医学
Journal of Neuro-Oncology Pub Date : 2025-03-06 DOI: 10.1007/s11060-025-04964-8
Joonas Laajava, Mika Niemelä, Miikka Korja
{"title":"Peritumoral edema resolves infrequently in surgically treated patients with intracranial meningioma- a retrospective study of 279 meningioma patients.","authors":"Joonas Laajava, Mika Niemelä, Miikka Korja","doi":"10.1007/s11060-025-04964-8","DOIUrl":"https://doi.org/10.1007/s11060-025-04964-8","url":null,"abstract":"<p><strong>Background: </strong>The resolution of peritumoral brain edema (PTBE) following surgery for intracranial meningioma (IM) is poorly understood. We hypothesized that PTBE represents a more permanent rather than resolving parenchymal change. Therefore, our aim was to assess the frequency of PTBE resolution following gross total resection (GTR) of IM.</p><p><strong>Methods: </strong>IM patients who were operated on in the study hospital between 2000 and 2020, who had preoperative magnetic resonance imaging (MRI) showing PTBE and a follow-up MRI performed at least one year after surgery, were retrospectively identified. To minimize confounding by PTBE related to a postoperative residual tumor, only patients who had undergone GTR were included. PTBE was defined as hyperintensity on either pre- or postoperative fluid-attenuated inversion recovery (FLAIR) MRI sequences.</p><p><strong>Results: </strong>A total of 279 adult meningioma patients were retrospectively identified. Of these, 208 (74.6%) were graded as World Health Organization grade 1 and 71 (25.4%) as grade 2. Of the 279 patients who had the first postoperative follow-up MRI at one year or later, PTBE changes persisted in 270 (96.8%) patients. However, over 90% resolution in PTBE volume was observed in 102 (35.8%) patients during the median MRI follow-up of 5.0 years (2.3-6.5). Higher edema index (p <.001) and temporal PTBE location (p =.018) were associated with higher resolution percentage of preoperative PTBE.</p><p><strong>Conclusion: </strong>Persisting PTBE is a common finding following GTR of IMs. While complete resolution of PTBE is rare, considerable resolution is often seen. The nature and exact cause of these persisting parenchymal changes are unclear, but they likely represent gliosis.</p>","PeriodicalId":16425,"journal":{"name":"Journal of Neuro-Oncology","volume":" ","pages":""},"PeriodicalIF":3.2,"publicationDate":"2025-03-06","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143567345","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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