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BACKGROUND: Acute encephalopathy with biphasic seizures and late reduced diffusion (AESD) develops along with status epilepticus and widespread subcortical white matter edema. We aimed to evaluate the epileptic foci and networks in two patients with epilepsy after AESD using simultaneous electroencephalography and functional magnetic resonance imaging (EEG-fMRI). METHODS: Statistically significant blood oxygen level-dependent (BOLD) responses related to interictal epileptiform discharges (IEDs) were analyzed using an event-related design of hemodynamic response functions with multiple peaks. RESULTS: Patient 1 developed focal seizures at age 10 years, one year after AESD onset. Positive BOLD changes were observed in the bilateral frontotemporal lobes, left parietal lobe, and left insula. BOLD changes were also observed in the subcortical structures. Patient 2 developed epileptic spasms at age two years, one month after AESD onset. Following total corpus callosotomy (CC) at age three years, the epileptic spasms resolved, and neurodevelopmental improvement was observed. Before CC, positive BOLD changes were observed bilaterally in the frontotemporal lobes. BOLD changes were also observed in the subcortical structures. After CC, the positive BOLD changes were localized in the temporal lobe ipsilateral to the IEDs, and the negative BOLD changes were mainly in the cortex and subcortical structures of the hemisphere ipsilateral to IEDs. CONCLUSION: EEG-fMRI revealed multiple epileptic foci and extensive epileptic networks, including subcortical structures in two cases with post-AESD epilepsy. CC may be effective in disconnecting the bilaterally synchronous epileptic networks of epileptic spasms after AESD, and pre-and post-operative changes in EEG-fMRI may reflect improvements in epileptic symptoms.

DOI： 10.1016/j.braindev.2024.07.003

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Authorship：Last author   Language：English   Publisher：Journal of Neuroscience Methods  

The authors regret that the affiliation information was incomplete in the original article. The correct and complete affiliation information should be as follows: Akihiro Mizunoa,ba Department of Neurosurgery, Komaki City Hospital, Aichi, Japan b Department of Neurosurgery, Nagoya University, Nagoya, Japan This research was conducted while the author was affiliated with Nagoya University. The addition of this affiliation is important for accurately reflecting the research context. The authors would like to apologize for any inconvenience caused.

DOI： 10.1016/j.jneumeth.2024.110293

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Language：English   Publisher：Brain Tumor Pathology  

High-grade supratentorial tumors harboring ZFTA::NCOA1/2 fusion in infants presenting with mixed histology of embryonal-appearing components resembling ependymoma and mesenchymal sarcomatous components have recently been reported as ependymoma-like tumors with mesenchymal differentiation (ELTMDs). In contrast, we describe herein a pathologically similar case with a novel ZFTA::RELA fusion in an adult. A frontal lobe lesion was resected from a 30-year-old woman and displayed mixed components on pathological examination, showing ependymoma-like and sarcomatous parts. The absence of perivascular pseudorosettes was inconsistent with a diagnosis of ependymoma. Fluorescence in situ hybridization analysis confirmed ZFTA::RELA fusion. The DKFZ methylation classifier (v12.8) did not categorize this case among established methylation classes. In addition, t-distributed stochastic neighbor embedding analysis using DNA methylation data revealed that the present case was distant from ependymomas but close to two previously reported cases of ELTMD involving ZFTA::NCOA1/2 fusion. Taken together, we concluded that this tumor should be considered under the entity of ELTMD. This represents the first description of an adult patient with ELTMD harboring ZFTA::RELA fusion analyzed by DNA methylation profiling, supporting the establishment of ELTMD as a possible new tumor type.

DOI： 10.1007/s10014-024-00489-6

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Language：English   Publisher：Japanese journal of clinical oncology  

BACKGROUND: This study aimed to investigate what treatment are selected for malignant brain tumors, particularly glioblastoma (GBM) and primary central nervous system lymphoma (PCNSL), in real-world Japan and the costs involved. METHODS: We conducted a questionnaire survey regarding treatment selections for newly diagnosed GBM and PCNSL treated between July 2021 and June 2022 among 47 institutions in the Japan Clinical Oncology Group-Brain Tumor Study Group. We calculated the total cost and cost per month of the initial therapy for newly diagnosed GBM or PCNSL. RESULTS: The most used regimen (46.8%) for GBM in patients aged ≤74 years was 'Surgery + radiotherapy concomitant with temozolomide'. This regimen's total cost was 7.50 million JPY (Japanese yen). Adding carmustine wafer implantation (used in 15.0%), TTFields (used in 14.1%), and bevacizumab (BEV) (used in 14.5%) to the standard treatment of GBM increased the cost by 1.24 million JPY for initial treatment, and 1.44 and 0.22 million JPY per month, respectively. Regarding PCNSL, 'Surgery (biopsy) + rituximab, methotrexate, procarbazine, and vincristine (R-MPV) therapy' was the most used regimen (42.5%) for patients of all ages. This regimen incurred 1.07 million JPY per month. The three PCNSL regimens based on R-MPV therapy were in ultra-high-cost medical care (exceeding 1 million JPY per month). CONCLUSIONS: Treatment of malignant brain tumors is generally expensive, and cost-ineffective treatments such as BEV are frequently used. We believe that the results of this study can be used to design future economic health studies examining the cost-effectiveness of malignant brain tumors.

DOI： 10.1093/jjco/hyae116

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BACKGROUND: Ependymal cilia play a major role in the circulation of cerebrospinal fluid. Although isolation of cilia is an essential technique for investigating ciliary structure, to the best of our knowledge, no report on the isolation and structural analysis of ependymal cilia from mouse brain is available. NEW METHOD: We developed a novel method for isolating ependymal cilia from mouse brain ventricles. We isolated ependymal cilia by partially opening the lateral ventricles and gently applying shear stress, followed by pipetting and ultracentrifugation. RESULTS: Using this new method, we were able to observe cilia separately. The results demonstrated that our method successfully isolated intact ependymal cilia with preserved morphology and ultrastructure. In this procedure, the ventricular ependymal cell layer was partially detached. COMPARISON WITH EXISTING METHODS: Compared to existing methods for isolating cilia from other tissues, our method is meticulously tailored for extracting ependymal cilia from the mouse brain. Designed with a keen understanding of the fragility of the ventricular ependyma, our method prioritizes minimizing tissue damage during the isolation procedure. CONCLUSIONS: We isolated ependymal cilia from mouse brain by applying shear stress selectively to the ventricles. Our method can be used to conduct more detailed studies on the structure of ependymal cilia.

DOI： 10.1016/j.jneumeth.2024.110198

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Authorship：Last author   Language：English   Publisher：The Japan Neurosurgical Society  

<p>Parent artery occlusion for large or giant internal carotid artery aneurysms remains a necessary procedure in the era of flow diverters. Endovascular parent artery occlusion is currently performed using detachable balloons or coils, which are difficult to obtain or costly. At our institution, we have devised a technique for combining n-butyl-2-cyanoacrylate and coils with flow control to solve this problem. Patients who underwent parent artery occlusion for large or giant internal carotid artery aneurysms with a follow-up period of more than 12 months were included. Imaging outcomes were evaluated for complete or incomplete aneurysmal occlusion and with or without aneurysmal shrinkage. The clinical outcome was based on changes in the modified Rankin Scale. Ten patients (ten aneurysms) were included. Their average age and average follow-up period were 68.4 years and 36 months, respectively. Complete occlusion and favorable clinical outcome were observed in all cases. The parent artery occlusion using a combination of coils and n-butyl-2-cyanoacrylate with flow control technique is effective for both imaging and clinical outcomes.</p>

DOI： 10.2176/jns-nmc.2024-0046

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DOI： 10.7759/cureus.67884

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PURPOSE: This study identified the factors affecting cerebral microbleed (CMBs) development. Moreover, their effects on intelligence and memory and association with stroke in patients with germinoma who had long-term follow-up were evaluated. METHODS: This study included 64 patients with germinoma who were histologically and clinically diagnosed with and treated for germinoma. These patients were evaluated cross-sectionally, with a focus on CMBs on susceptibility-weighted magnetic resonance imaging (SWI), brain atrophy assessed through volumetric analysis, and intelligence and memory. RESULTS: The follow-up period was from 32 to 412 (median: 175.5) months. In total, 43 (67%) patients had 509 CMBs and 21 did not have CMBs. Moderate correlations were observed between the number of CMBs and time from initial treatments and recurrence was found to be a risk factor for CMB development. Increased temporal CMBs had a marginal effect on the processing speed and visual memory, whereas brain atrophy had a statistically significant effect on verbal, visual, and general memory and a marginal effect on processing speed. Before SWI acquisition and during the follow-up periods, eight strokes occurred in four patients. All of these patients had ≥ 15 CMBs on SWI before stroke onset. Meanwhile, 33 patients with < 14 CMBs or 21 patients without CMBs did not experience stroke. CONCLUSION: Patients with a longer time from treatment initiation had a higher number of CMBs, and recurrence was a significant risk factor for CMB development. Furthermore, brain atrophy had a stronger effect on memory than CMBs. Increased CMBs predict the stroke onset.

DOI： 10.1007/s11060-024-04753-9

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Language：Japanese   Publisher：一般社団法人 日本内分泌学会  

DOI： 10.1507/endocrine.100.s.hpt_19

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Authorship：Last author   Language：English   Publisher：BMC Cancer  

Background: Stereotactic irradiation has become the mainstay treatment for brain metastases (BM), and whole-brain radiotherapy (WBRT) is often used for symptom palliation. However, the survival time of patients with BM undergoing palliative WBRT (pWBRT) is limited, making it difficult to select patients who should receive treatment. Methods: We collected patient data from 2016 to 2022 at the Shizuoka Cancer Center and retrospectively analyzed the factors related to survival time. Overall survival (OS) was defined as the survival time after WBRT. Results: A total of 301 patients (median age, 66 years) who underwent pWBRT were included. The primary cancers were lung, breast, gastrointestinal tract, and other cancers in 203 (67%), 38 (13%), 33 (11%), and 27 (9%) patients, respectively. Median OS of all patients was 4.1 months. In the multivariate analysis, male sex (hazard ratio [HR]:1.4), Karnofsky Performance Status (KPS) ≤ 60 (HR:1.7), presence of extracranial metastasis (ECM) (HR:1.6), neutrophil-lymphocyte ratio (NLR) ≥ 5 (HR:1.6), and lactate dehydrogenase (LDH) ≥ upper limit of normal (ULN) (HR:1.3) were significantly associated with shorter OS (all P < 0.05). To predict the OS, we created a prognostic scoring system (PSS). We gave one point to each independent prognostic factor. Median OS for patients with scores of 0–2, 3, and 4–5 were 9.0, 3.5 and 1.7 months, respectively (P < 0.001). Conclusions: Male sex, KPS ≤ 60, presence of ECM, NLR ≥ 5, and LDH ≥ ULN were poor prognostic factors for patients with BM undergoing pWBRT. By PSS combining these factors, it may be possible to select patients who should undergo pWBRT.

DOI： 10.1186/s12885-024-12729-1

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BACKGROUND/AIM: The prognosis of patients with brain metastases (BMs) originating from lung cancer remains poor, despite advancements in treatment strategies. The role of tertiary lymphoid structures (TLSs) within the tumor immune microenvironment of BMs has not been extensively explored. PATIENTS AND METHODS: This study utilized patient-derived clinical samples from 17 patients with histologically confirmed BMs of lung cancer, undergoing surgical resection. Immunohistochemistry was employed to analyze the presence and characteristics of TLS and tumor-infiltrating lymphocytes (TILs) within BM tissues, correlating these with clinical outcomes. RESULTS: TLSs, albeit in their immature form, were identified within BM tissues, distinguishing them from their mature counterparts in primary lung cancer tissues. A significant correlation between TLS density (but not TIL density) and improved postoperative survival was observed, underscoring the potential of TLS density as an independent prognostic marker. Furthermore, TLS density did not correlate with the Graded Prognostic Assessment (GPA) index, suggesting its unique prognostic value beyond conventional predictors. CONCLUSION: Our findings reveal the presence of TLSs in lung cancer-derived BMs and highlight their prognostic significance, independent of the GPA index. The identification of TLS within the unique central nervous system tumor microenvironment offers new insights into the immune landscape of BMs and suggests potential avenues for immunotherapeutic interventions targeting these structures to improve patient outcomes.

DOI： 10.21873/anticanres.17184

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Language：English   Publishing type：Research paper (scientific journal)   Publisher：Springer Science and Business Media LLC  

DOI： 10.1007/s11060-024-04687-2

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Other Link： https://link.springer.com/article/10.1007/s11060-024-04687-2/fulltext.html

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DOI： 10.7759/cureus.65547

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DOI： 10.1227/neu.0000000000003115

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Authorship：Last author   Language：English   Publisher：Child's Nervous System  

Purpose: Filum terminale lipoma (FTL) causes spinal-cord tethering and is associated with tethered-cord syndrome, which is treated by dissection of the entrapment. The conventional treatment for FTL involves dissection of the spinal cord through a laminotomy open approach (LOA). However, in recent years, the interlaminar approach (ILA) has gained popularity as a minimally invasive surgery. This study compares the effectiveness of the minimally invasive ILA with the conventional LOA in treating FTL. Methods: We retrospectively evaluated data on the ILA and LOA for FTL at our center. In total, 103 participants were enrolled, including 55 in the ILA group and 48 in the LOA group. Results: The ILA required significantly less surgical time and resulted in less blood loss. The improvement rate of symptoms in symptomatic patients was 84%, and for urinary symptoms and abnormal urodynamic study findings, it was 77%. The postoperative maintenance rate for asymptomatic patients was 100%. Postoperative complications of ILA included delayed wound healing in two patients (3.6%). Conclusion: Compared with LOA, ILA offers advantages in terms of shorter operative time and less blood loss, with no significant difference in long-term symptom-improvement rates between the groups.

DOI： 10.1007/s00381-024-06507-4

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OBJECTIVE: Transient neurological deficits (TNDs) are known to develop after direct bypass for moyamoya disease (MMD) and may be risk factors for subsequent stroke. However, the factors involved in the development of TNDs and stroke after indirect revascularization alone, including their association with subsequent stroke, remain unclear. The purpose of this study was to investigate this issue. METHODS: The subjects of the study were 30 patients with MMD who underwent a total of 40 indirect revascularization procedures at our institution. Clinical and radiological data were collected retrospectively. To examine factors associated with the development of postoperative TND/stroke/asymptomatic disease, the clinical characteristics of each group were statistically compared. RESULTS: The mean age at surgery was 7 years (range 1-63). TNDs developed after surgery in 9 out of 40 patients (22.5%). Stroke in the acute postoperative period occurred in 3 patients (7.5%), all of whom experienced cerebral infarctions. Demographic data and preoperative clinical information were not different between the groups. However, posterior cerebral artery (PCA) involvement on preoperative imaging was significantly associated with the development of TNDs and stroke (p=0.006). Furthermore, postoperative stroke was associated with unfavorable outcomes (p=0.025). CONCLUSIONS: PCA involvement is significantly associated with the occurrence of TNDs. In contrast, TNDs after indirect revascularization have little relationship with the subsequent development of stroke. TNDs usually resolve without new strokes, and a better understanding of this particular pathology could help establish an optimal treatment regimen.

DOI： 10.1016/j.wneu.2024.04.135

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Authorship：Last author,　Corresponding author   Language：English   Publisher：The Japan Neurosurgical Society  

<p>Brain tumor-related epilepsy (BTRE) is a complication that significantly impairs the quality of life and course of treatment of patients with brain tumors. Several recent studies have shed further light on the mechanisms and pathways by which genes and biological molecules in the tumor microenvironment can cause epilepsy. Moreover, epileptic seizures have been found to promote the growth of brain tumors, making the control of epilepsy a critical factor in treating brain tumors. In this study, we summarize the previous research and recent findings concerning BTRE. Expectedly, a deeper understanding of the underlying genetic and molecular mechanisms leads to safer and more effective treatments for suppressing epileptic symptoms and tumor growth.</p>

DOI： 10.2176/jns-nmc.2023-0299

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Language：English   Publisher：Parkinsonism and Related Disorders  

Introduction: Parkinson's disease (PD) presents with decreased heart rate variability (HRV) from its early stages. However, most of its evidence originates from HRV measurements in parasympathetic dominant states. In this study, we aimed to examine whether HRV in sympathetic dominant states during the head-up tilt table test (HUT) serves as a marker of autonomic dysfunction in PD and isolated REM sleep behavior disorder (iRBD). Methods: We retrospectively assessed 102 patients with PD, 10 patients with iRBD, and 43 healthy controls. We then measured the coefficient of variation of RR intervals as an HRV parameter in sympathetic dominant states (CVRR-S) and parasympathetic dominant states (CVRR-P). Furthermore, we evaluated parameters of cardiac autonomic function, including HUT and the heart-to-mediastinum (H/M) ratio of cardiac metaiodobenzylguanidine scintigraphy. Results: Patients with iRBD and PD at Hoehn and Yahr stage I exhibited a significantly decreased CVRR-S compared to healthy controls (controls vs. iRBD vs. PD; 1.82 ± 0.64 % vs. 1.13 ± 0.41 % vs. 1.15 ± 0.51 %, p < 0.001), although no further deterioration was observed in PD at more severe Hoehn and Yahr stages. CVRR-S showed a significant correlation with the H/M ratio in PD (r = 0.51, p < 0.001). Additionally, receiver operating characteristic (ROC) analysis revealed a larger area under the ROC curve in CVRR-S compared to that in CVRR-P for discriminating PD or iRBD from healthy controls. Conclusion: HRV in sympathetic dominant states shows the potential to be a marker of autonomic dysfunction in iRBD and early-stage PD, aiding in early diagnosis and patient stratification.

DOI： 10.1016/j.parkreldis.2024.107020

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Authorship：Last author   Language：Japanese   Publisher：株式会社医学書院  

DOI： 10.11477/mf.1436204969

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BACKGROUND: Posterior clinoid process (PCP) meningioma is an exceedingly rare entity. It remains the most challenging skull base lesion for neurosurgeons due to its treacherous location that insinuates amongst critical neurovascular structures. This article will describe the technical notes using the endoscopic endonasal approach that provide the earliest devascularization and detachment of the tumor PCP meningioma. METHODS: We are introducing the surgical implementation of an endoscopic endonasal approach to removing PCP meningioma. Furthermore, we perform a literature review of posterior clinoid process meningioma that undergoes surgical intervention, then summarize the benefits and limitations of each approach. RESULTS: We present a case of right PCP meningioma that was removed using an endoscopic endonasal approach through the transposterior clinoid corridor in a 52-year-old-woman. We describe the technical notes in performing this approach to have the earliest devascularization and detachment of the tumor by performing posterior clinoidectomy. Safe tumor removal is performed with a wide and clear view of the surrounding neurovascular structure. Based on our database search, we found nine articles reported on the surgical management of PCP meningiomas, with a total number of 15 cases. All of the reported cases performed the tumor removal using the transcranial approach. CONCLUSION: The endoscopic endonasal transposterior clinoid approach circumvents all disadvantages faced by the traditional transcranial approach, providing the earliest approach to devascularized and detaching the tumor from its attachment at PCP. This approach demonstrates safety and efficacy, making it an acceptable alternative for PCP meningioma resections.

DOI： 10.3389/fonc.2024.1368277

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Publisher：三輪書店  

DOI： 10.11477/mf.5002202293

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Authorship：Last author   Language：English   Publishing type：Research paper (scientific journal)   Publisher：Journal of Neurotrauma  

Spinal cord injury (SCI) induces devastating permanent deficits. Recently, cell transplantation therapy has become a notable treatment for SCI. Although stem cells from human exfoliated deciduous teeth (SHED) are an attractive therapy, their precise mechanism of action remains to be elucidated. In this study, we explored one of the neuroprotective mechanisms of SHED treatment at the subacute stage after SCI. We used a rat clip compression SCI model. The animals were randomly divided into three groups: SCI, SCI + phosphate-buffered saline (PBS), and SCI + SHED. The SHED or PBS intramedullary injection was administered immediately after SCI. After SCI, we explored the effects of SHED on motor function, as assessed by the Basso-Beattie-Bresnahan score and the inclined plane method, the signal transduction pathway, especially the Janus kinase (JAK) and the signal transducer and activator of transcription 3 (STAT3) pathway, the apoptotic pathway, and the expression of neurocan, one of the chondroitin sulfate proteoglycans. SHED treatment significantly improved functional recovery from Day 14 relative to the controls. Western blot analysis showed that SHED significantly reduced the expression of glial fibrillary acidic protein (GFAP) and phosphorylated STAT3 (p-STAT3) at Tyr705 on Day 10 but not on Day 5. However, SHED had no effect on the expression levels of Iba-1 on Days 5 or 10. Immunohistochemistry revealed that p-STAT3 at Tyr705 was mainly expressed in GFAP-positive astrocytes on Day 10 after SCI, and its expression was reduced by administration of SHED. Moreover, SHED treatment significantly induced expression of cleaved caspase 3 in GFAP-positive astrocytes only in the epicenter lesions on Day 10 after SCI but not on Day 5. The expression of neurocan was also significantly reduced by SHED injection on Day 10 after SCI. Our results show that SHED plays an important role in reducing astrogliosis and glial scar formation between Days 5 and 10 after SCI, possibly via apoptosis of astrocytes, ultimately resulting in improvement in neurological functions thereafter. Our data revealed one of the neuroprotective mechanisms of SHED at the subacute stage after SCI, which improved functional recovery after SCI, a serious condition.

DOI： 10.1089/neu.2023.0251

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Radiotherapy combined with temozolomide (TMZ+RT) is the primary treatment for high-grade glioma. TMZ is classified as a moderate emetic risk agent and, thus, supportive care for nausea and vomiting is important. In Nagoya University Hospital, all patients are treated with a 5-hydroxy-tryptamine 3 receptor antagonist (5-HT3RA) for the first 3 days. The daily administration of 5-HT3RA is resumed after the 4th day based on the condition of patients during TMZ+RT. Therefore, the present study investigated risk factors for nausea and vomiting in patients requiring the daily administration of 5-HT3RA. Patients with high-grade glioma who received TMZ+RT between January 2014 and December 2019 at our hospital were included. Patients were divided into two groups: a control group (patients who did not resume 5-HT3RA) and resuming 5-HT3RA group (patients who resumed 5-HT3RA after the 4th day), and both groups were compared to identify risk factors for nausea and vomiting during TMZ+RT. There were 78 patients in the control group (68%) and 36 in the resuming 5-HT3RA group (32%). A multivariate analysis of patient backgrounds in the two groups identified age <18 years, PS 2 or more, and occipital lobe tumors as risk factors for nausea and vomiting. Nausea and vomiting were attenuated in 30 patients (83%) in the resuming 5-HT3RA group following the resumption of 5-HT3RA. The results obtained highlight the importance of extracting patients with these risk factors before the initiation of therapy and the early resumption or daily administration of 5-HT3RA according to the condition of each patient.

DOI： 10.18999/nagjms.86.2.304

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Authorship：Last author   Language：English   Publishing type：Research paper (scientific journal)   Publisher：World Neurosurgery  

Objective: Intracranial dural arteriovenous fistulae (DAVFs) represent a subset of cerebral vascular malformations associated with significant morbidity and mortality. In Japan, DAVF exhibits sex-based differences in anatomical distribution, with female predominance in the cavernous sinus (CS) and male predominance in the transverse sinus (TS). Nevertheless, the pathophysiology of DAVF is not fully understood, and hormonal influences are hypothesized to play a role in its development. This study aimed to investigate changes in the concentrations of sex steroid hormones between intracranial and peripheral sampling sites in patients with CS- and TS-DAVF. Methods: We recruited 19 patients with CS-DAVF (n = 12) and TS-DAVF (n = 7) in this study. Blood hormone measurements were obtained from peripheral and jugular bulb samples during endovascular intervention. Hormone concentrations were analyzed using enzyme-linked immunosorbent assay kits, and statistical analyses were performed. Results: Our study revealed a higher prevalence of CS-DAVF in females and TS-DAVF in males, which is consistent with previous studies. Estradiol concentration was significantly lower in the jugular bulb compared with in the periphery in both patients with CS- and TS-DAVF. This decrease in estradiol was observed irrespective of the patient's sex and independent of follicle-stimulating hormone levels. Conclusions: These findings indicate a local decrease in estradiol levels within the intracranial vasculature of patients with DAVF. This suggests a potential multifactorial role of estradiol in the pathomechanism of DAVFs, warranting further investigation to understand its influence on DAVF formation and potential targeted therapies, thereby enhancing patient outcomes.

DOI： 10.1016/J.WNEu.2024.02.052

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Neurofibromatosis type 1 (NF1) is an autosomal dominant syndrome caused by germline alteration of the NF1gene. Among various NF1-related manifestations, obstructive hydrocephalus especially in adult NF1 cases is less frequently found. We report two adult NF1 cases exhibiting obstructive hydrocephalus due to an aggressive posterior fossa tumor exhibiting pathological characteristics of pilocytic astrocytoma as NF1-related manifestations. In these two cases, we performed endoscopic third ventriculostomy (ETV) and tumor biopsy as an initial treatment. The initial pathological diagnosis of the tumor is conventional pilocytic astrocytoma. After biopsy both cases revealed rapid tumor growth, therefore, we performed tumor removal, chemotherapy, and radiation therapy during an aggressive clinical course. However, both cases revealed dismal prognosis due to the progression of the tumor in spite of successful management of hydrocephalus by an initial ETV. DNA methylation analysis revealed that the tumor of one case matched high-grade astrocytoma with piloid features (HGAP). Most central nervous system tumors developed in NF1 are less aggressive such as pilocytic astrocytoma; however, recently a few studies revealed that HGAP, which has been a newly introduced malignant tumor in the World Health Organization Classification of Tumors of the Central Nervous System, 5th edition (WHO CNS 5), rarely develops in NF1 cases. These findings suggested that HGAP might be one of the important causes of obstructive hydrocephalus in adult NF1 cases.

DOI： 10.7759/cureus.58697

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Li-Fraumeni syndrome (LFS) is an autosomal dominant tumor predisposition syndrome caused by heterozygous germline mutations or deletions in the TP53 tumor suppressor gene. Central nervous system tumors, such as choroid plexus tumors, medulloblastomas, and diffuse gliomas, are frequently found in patients with LFS. Although molecular profiles of diffuse gliomas that develop in pediatric patients with LFS have been elucidated, those in adults are limited. Recently, diffuse gliomas have been divided into pediatric- and adult-type gliomas, based on their distinct molecular profiles. In the present study, we investigated the molecular profiles of high-grade gliomas in three adults with LFS. These tumors revealed characteristic histopathological findings of high-grade glioma or glioblastoma and harbored wild-type IDH1/2 according to whole exome sequencing (WES). However, these tumors did not exhibit the key molecular alterations of glioblastoma, IDH-wildtype such as TERT promoter mutation, EGFR amplification, or chromosome 7 gain and 10 loss. Although WES revealed no other characteristic gene mutations or copy number alterations in high-grade gliomas, such as those in histone H3 genes, PDGFRA amplification was found in all three cases together with uniparental disomy of chromosome 17p, where the TP53 gene is located. DNA methylation analyses revealed that all tumors exhibited DNA methylation profiles similar to those of pediatric-type high-grade glioma H3-wildtype and IDH-wildtype (pHGG H3-/IDH-wt), RTK1 subtype. These data suggest that high-grade gliomas developed in adult patients with LFS may be involved in pHGG H3-/IDH-wt. PDGFRA and homozygous alterations in TP53 may play pivotal roles in the development of this type of glioma in adult patients with LFS.

DOI： 10.1186/s40478-024-01762-7

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A prompt and reliable molecular diagnosis for brain tumors has become crucial in precision medicine. While Comprehensive Genomic Profiling (CGP) has become feasible, there remains room for enhancement in brain tumor diagnosis due to the partial lack of essential genes and limitations in broad copy number analysis. In addition, the long turnaround time of commercially available CGPs poses an additional obstacle to the timely implementation of results in clinics. To address these challenges, we developed a CGP encompassing 113 genes, genome-wide copy number changes, and MGMT promoter methylation. Our CGP incorporates not only diagnostic genes but also supplementary genes valuable for research. Our CGP enables us to simultaneous identification of mutations, gene fusions, focal and broad copy number alterations, and MGMT promoter methylation status, with results delivered within a minimum of 4 days. Validation of our CGP, through comparisons with whole-genome sequencing, RNA sequencing, and pyrosequencing, has certified its accuracy and reliability. We applied our CGP for 23 consecutive cases of intracranial mass lesions, which demonstrated its efficacy in aiding diagnosis and prognostication. Our CGP offers a comprehensive and rapid molecular profiling for gliomas, which could potentially apply to clinical practices and research primarily in the field of brain tumors.

DOI： 10.1007/s10014-023-00476-3

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Language：Japanese   Publisher：(一社)日本小児神経外科学会  

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Authorship：Last author   Language：English   Publishing type：Research paper (scientific journal)   Publisher：Parkinsonism and Related Disorders  

Introduction: Connector hubs are specialized brain regions that connect multiple brain networks and therefore have the potential to affect the functions of multiple systems. This study aims to examine the involvement of connector hub regions in essential tremor. Methods: We examined whole-brain functional connectivity alterations across multiple brain networks in 27 patients with essential tremor and 27 age- and sex-matched healthy controls to identify affected hub regions using a network metric called functional connectivity overlap ratio estimated from resting-state functional MRI. We also evaluated the relationships of affected hubs with cognitive and tremor scores in all patients and with motor function improvement scores in 15 patients who underwent postoperative follow-up evaluations after focused ultrasound thalamotomy. Results: We have identified affected connector hubs in the cerebellum and thalamus. Specifically, the dentate nucleus in the cerebellum and the dorsomedial thalamus exhibited more extensive connections with the sensorimotor network in patients. Moreover, the connections of the thalamic pulvinar with the visual network were also significantly widespread in the patient group. The connections of these connector hub regions with cognitive networks were negatively associated (FDR q < 0.05) with cognitive, tremor, and motor function improvement scores. Conclusion: In patients with essential tremor, connector hub regions within the cerebellum and thalamus exhibited widespread functional connections with sensorimotor and visual networks, leading to alternative pathways outside the classical tremor axis. Their connections with cognitive networks also affect patients’ cognitive function.

DOI： 10.1016/j.parkreldis.2024.106034

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Authorship：Last author   Language：English   Publisher：The Japan Neurosurgical Society  

<p>Magnetic resonance-guided focused ultrasound (MRgFUS) thalamotomy is an effective treatment for essential tremor (ET). However, its long-term outcomes and prognostic factors remain unclear. This study aimed to retrospectively investigate 38 patients with ET who underwent MRgFUS thalamotomy and were followed up for >2 years. The improvement in tremor was evaluated using the Clinical Rating Scale for Tremor (CRST). Adverse events were documented, and correlations with factors, such as skull density ratio (SDR), maximum mean temperature (T-max), and lesion size, were examined. Furthermore, the outcomes were compared between two groups, one that met the cutoff values, which was previously reported (preoperative CRST-B ≤ 25, T-max ≥ 52.5°C, anterior-posterior size of lesion ≥ 3.9 mm, superior-inferior [SI] size of lesion > 5.5 mm), and the other that did not. The improvement rate was 59.4% on average at the 2-year follow-up. Adverse events, such as numbness (15.8%), dysarthria (10.5%), and lower extremity weakness (2.6%), were observed even after 2 years, although these were mild. The factors correlated with tremor improvement were the T-max and SI size of the lesion (p < 0.05), whereas the SDR showed no significance. Patients who met the aforementioned cutoff values demonstrated a 69.8% improvement at the 2-year follow-up, whereas others showed a 43.6% improvement (p < 0.05). In conclusion, MRgFUS is effective even after 2 years. The higher the T-max and the larger the lesion size, the better the tremor control. Previously reported cutoff values clearly predict the 2-year prognosis, indicating the usefulness of MRgFUS.</p>

DOI： 10.2176/jns-nmc.2023-0202

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Authorship：Last author   Language：English   Publishing type：Research paper (scientific journal)   Publisher：Epilepsia Open  

Objective: Patients with epilepsy have high risk of experiencing uncommon causes of death. This study aimed to evaluate patients who underwent unusual deaths related to epilepsy and identify factors that may contribute to these deaths and may also include sudden unexpected death in epilepsy (SUDEP). Methods: We analyzed 5291 cases in which a postmortem imaging (PMI) study was performed using plane CT, because of an unexplained death. A rapid troponin T assay was performed using peripheral blood samples. Clinical information including the cause of death suspected by the attending physician, body position, place of death, medical history, and antiseizure medications was evaluated. Results: A total of 132 (2.6%) patients had an obvious history of epilepsy, while 5159 individuals had no history of epilepsy (97.4%). Cerebrovascular disease was the cause of death in 1.6% of patients in the group with epilepsy, and this was significantly lower than that in the non-epilepsy group. However, drowning was significantly higher (9.1% vs. 4.4%). Unspecified cause of death was significantly more frequent in the epilepsy group (78.0% vs. 57.8%). Furthermore, the proportion of patients who demonstrated elevation of troponin T levels without prior cardiac disease was significantly higher in the epilepsy group (37.9% vs. 31.1%). At discovery of death, prone position was dominant (30.3%), with deaths occurring most commonly in the bedroom (49.2%). No antiseizure medication had been prescribed in 12% of cases, while 29.5% of patients were taking multiple antiseizure medications. Significance: The prevalence of epilepsy in individuals experiencing unusual death was higher than in the general population. Despite PMI studies, no definitive cause of death was identified in a significant proportion of cases. The high troponin T levels may be explained by long intervals between death and examination or by higher incidence of myocardial damage at the time of death. Plain Language Summary: This study investigated unusual deaths in epilepsy patients, analyzing 5291 postmortem imaging cases. The results showed that 132 cases (2.6%) had a clear history of epilepsy. In these cases, only 22% cases were explained after postmortem examination, which is less than in non-epilepsy group (42.2%). Cerebrovascular disease was less common in the epilepsy group, while drowning was more common. Elevated troponin T levels, which suggest possibility of myocardial damage or long intervals between death and examination, were also more frequent in the epilepsy group compared to non-epilepsy group.

DOI： 10.1002/epi4.12891

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Authorship：Last author   Language：English   Publisher：Acta Neurochirurgica  

Background: In recent years, molecular findings on spinal gliomas have become increasingly important. This study aimed to investigate the role of 18F-fluorodeoxyglucose positron emission tomography/computed tomography (18F-FDG-PET/CT) in the diagnosis of spinal glioma. Methods: This study included patients diagnosed with spinal cord glioma who underwent 18F-FDG-PET examination at the Department of Neurosurgery, Nagoya University Hospital between January 2016 and November 2023. The gliomas were divided into two groups, high-grade and low-grade, based on pathological and molecular studies. The maximum standardized uptake values (SUVmax) of the tumors were quantified and subsequently represented using receiver operating characteristic (ROC) curves. Results: Eighteen participants were included in this study. Of the participants, seven had high-grade glioma with an SUVmax of 6.76 ± 0.72, and eleven had low-grade glioma with an SUVmax of 4.02 ± 1.78, and a statistically significant difference between the two groups. The ROC curve delineated an SUVmax cutoff value of 5.650, with an area under the curve (AUC) of approximately 0.909. Based on the cutoff value, the results of the diagnostic performance rendered a sensitivity and negative predictive value of 1.0, whereas the specificity and positive predictive value were 0.909 and 0.875, respectively. Conclusions: The present study shows that 18F-FDG-PET exhibits a markedly sensitive and negative predictive value in the assessment of spinal gliomas. Additionally, these findings have potential implications for the qualitative assessment of spinal gliomas using 18F-FDG-PET/CT. This imaging modality may be useful for making timely treatment decisions in situations where a detailed diagnosis by molecular analysis is not possible.

DOI： 10.1007/s00701-024-06039-5

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Chiari III malformation, a rare and severe subtype of Chiari malformations, is frequently associated with hydrocephalus. The conventional treatment approaches for hydrocephalus in Chiari III malformations have mainly focused on ventriculoperitoneal (VP) shunting, but optimal methods and timing remain uncertain. We report a case of a newborn girl with Chiari III malformation who underwent surgical closure of an occipitocervical encephalocele and ventricular reservoir implantation on her third day of life. This procedure successfully managed her hydrocephalus without significant short-term complications. Three months post-surgery, she developed secondary external hydrocephalus, which was managed through subdural-peritoneal shunting, allowing her to thrive until at least five years of age. This case demonstrates the potential of ventricular reservoir implantation in treating hydrocephalus associated with Chiari III malformation and brings to light secondary external hydrocephalus, subsequently managed by VP shunting.

DOI： 10.7759/cureus.55896

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This report examines delayed leukoencephalopathy as a postoperative complication after the use of flow diverter (FD) devices for endovascular cerebral aneurysm treatment. A case involving a 78-year-old female treated with a pipeline embolization device for a left internal carotid artery aneurysm is presented. Despite adherence to dual anti-platelet therapy, the patient developed intermittent headaches and memory issues 3 months post-operation. MRI revealed T1-enhancing foci and T2 hyperintense signal abnormalities in the left cerebral hemisphere, without new ischemic lesions, indicating potential embolic events or foreign body reactions. Following aphasia, a change from clopidogrel to prasugrel and the initiation of steroid pulse therapy led to the resolution of symptoms and MRI abnormalities over 6 months. This case underscores the reversibility of delayed leukoencephalopathy with appropriate intervention.

DOI： 10.1007/s00234-024-03281-7

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Language：English   Publisher：Cancers  

Awake surgery has become a standard practice for managing diffuse low-grade gliomas (LGGs), particularly in eloquent brain areas, and is established as a gold standard technique for left-dominant-hemisphere tumors. However, the intraoperative monitoring of functions in the right non-dominant hemisphere (RndH) is often neglected, highlighting the need for a better understanding of neurocognitive testing for complex functions in the right hemisphere. This article aims to comprehensively review the current literature on the benefits of awake craniotomy in gliomas of the non-dominant right hemisphere. A systematic review was conducted using the PubMed and ScienceDirect databases with keywords such as “right hemisphere”, “awake surgery”, “direct electrical brain stimulation and mapping”, and “glioma”. The search focused on anatomical and surgical aspects, including indications, tools, and techniques of awake surgery in right cerebral hemisphere gliomas. The literature search identified 74 sources, including original articles, books, monographs, and review articles. Two papers reported large series of language assessment cases in 246 patients undergoing awake surgery with detailed neurological semiology and mapping techniques, while the remaining studies were predominantly neuroradiological and neuroimaging in nature. Awake craniotomy for non-dominant-hemisphere gliomas is an essential tool. The term “non-dominant” should be revised, as this hemisphere contributes significantly to essential cognitive functions in the human brain.

DOI： 10.3390/cancers16061161

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Authorship：Corresponding author   Language：English   Publishing type：Research paper (scientific journal)  

BACKGROUND: Cerebral venous sinus thrombosis (CVST) sometimes occurs in the background of hypercoagulopathic disorders, including malignancy, chemotherapy, etc. Glioblastoma (GBM) is a malignancy found in the central nervous system, and reports on cases of GBM complicated by CVST are sparse. The authors herein report a case of GBM complicated by CVST during maintenance temozolomide (TMZ) chemotherapy and describe the utility of diffusion-weighted magnetic resonance imaging (MRI) for the detection of CVST. OBSERVATIONS: A 65-year-old male was treated for left temporal GBM. After surgical removal of the lesion, the patient was treated with chemoradiation therapy, which included 60 Gy local radiation with concomitant TMZ chemotherapy. He was subsequently received TMZ maintenance therapy. Routine MRI performed 7 months after surgery revealed no evidence of tumor recurrence. However, diffusion-weighted imaging (DWI) revealed a high-intensity signal at the posterior portion of the superior sagittal sinus, indicating the presence of a thrombus. In addition to the preexisting symptoms, the patient experienced some disorientation. Angiography revealed an obstruction in the superior sagittal sinus, right transverse sinus, right sigmoid sinus, and straight sinus. His symptoms improved with endovascular and anticoagulant therapy. LESSONS: Performing DWI during routine follow-up can help in the early diagnosis of CVST in patients with malignant gliomas.

DOI： 10.3171/CASE23762

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BACKGROUND: Distant recurrence can occur by infiltration along white matter tracts or dissemination through the cerebrospinal fluid (CSF). This study aimed to clarify the clinical features and mechanisms of recurrence in the dentate nucleus (DN) in patients with supratentorial gliomas. Based on the review of our patients, we verified the hypothesis that distant DN recurrence from a supratentorial lesion occurs through the dentato-rubro-thalamo-cortical (DRTC) pathway. METHODS: A total of 380 patients with supratentorial astrocytoma, isocitrate dehydrogenase (IDH)-mutant (astrocytoma), oligodendroglioma, IDH mutant and 1p/19q-codeleted (oligodendroglioma), glioblastoma, IDH-wild type (GB), and thalamic diffuse midline glioma, H3 K27-altered (DMG), who underwent tumor resection at our department from 2009 to 2022 were included in this study. Recurrence patterns were reviewed. Additionally, clinical features and magnetic resonance imaging findings before treatment, at the appearance of an abnormal signal, and at further progression due to delayed diagnosis or after salvage treatment of cases with recurrence in the DN were reviewed. RESULTS: Of the 380 patients, 8 (2.1%) had first recurrence in the DN, 3 were asymptomatic when abnormal signals appeared, and 5 were diagnosed within one month after the onset of symptoms. Recurrence in the DN developed in 8 (7.4%) of 108 cases of astrocytoma, GB, or DMG at the frontal lobe or thalamus, whereas no other histological types or sites showed recurrence in the DN. At the time of the appearance of abnormal signals, a diffuse lesion developed at the hilus of the DN. The patterns of further progression showed that the lesions extended to the superior cerebellar peduncle, tectum, tegmentum, red nucleus, thalamus, and internal capsule along the DRTC pathway. CONCLUSION: Distant recurrence along the DRTC pathway is not rare in astrocytomas, GB, or DMG at the frontal lobe or thalamus. Recurrence in the DN developed as a result of the infiltration of tumor cells through the DRTC pathway, not dissemination through the CSF.

DOI： 10.1007/s00701-024-05981-8

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OBJECTIVE: Surgical treatment of brainstem cavernous malformations (CMs) is challenging. Surgery using the endoscopic transsphenoidal transclival approach (eTSTCA) is reported as a useful alternative for ventral brainstem CMs. However, CMs located in the ventral midline of the brainstem are rare, and only a small number of case reports on these CMs treated with the eTSTCA exist. The efficacy and safety of the eTSTCA have not yet been fully examined. METHODS: A retrospective analysis was performed for 5 consecutive patients who underwent surgery via the eTSTCA for treating ventral pontine CMs. RESULTS: The average maximum CM diameter was 26.0 mm (18-38 mm). All patients underwent MR-diffusion tensor imaging, which confirmed that the corticospinal tract (CST) deviated posteriorly or laterally to the CM. Direct brainstem cortical stimulation was performed to localize the CST before making the cortical incision. After the excision of the CM, the cavity was filled with artificial CSF to make an aqueous surgical field (wet-field technique) for observing the tumor cavity and confirming complete hemostasis and resection. Total removal was achieved in all patients. The preoperative modified Rankin Scale score was 3 in 3 patients and 4 in 2 patients, whereas it was 1 in 2 patients and 0 in 3 patients 3 months after surgery. Postoperative CSF leakage was observed in 1 patient, and transient abducens nerve palsy was observed in 1 patient. No other intra- or postoperative complications were observed. CONCLUSIONS: MR-diffusion tensor imaging and direct brainstem cortical stimulation were useful to ascertain the proximity of the CST to the CM. The endoscope provides a clear view even underwater, and it was safe and effective to observe the entire CM cavity and confirm complete hemostasis without additional retraction of the brainstem parenchyma, including the CST. The eTSTCA provides a direct access point to the lesion and may be a safer alternative treatment for patients whose CST deviates laterally or posteriorly to the CM.

DOI： 10.3171/2023.6.JNS23324

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Authorship：Last author   Language：English   Publisher：Nagoya Journal of Medical Science  

For patients with moyamoya disease, antiplatelet agents are often used during the perioperative periods of revascularization surgeries to prevent ischemic events. However, antiplatelet therapy is associated with the risk of hemorrhagic complications. Further, the influence of antiplatelet therapy on perioperative ischemic or hemorrhagic complications has not been investigated. This study aimed to determine the impact of antiplatelet agents on adult moyamoya disease patients with ischemic onset during the perioperative period. From January 2016 to December 2020, 183 consecutive combined (direct and indirect) revascularization surgeries for moyamoya disease patients were performed. Among these surgeries, 96 consecutive combined revascularization surgeries for adult moyamoya disease patients with ischemic onset were analyzed and perioperative ischemic and hemorrhagic complications were reviewed. Antiplatelet agents were continued during the perioperative period including on the day of surgery and the day after the surgery. Among 96 surgeries, no hemorrhagic complications occurred postoperatively. Infarction occurred in five cases (5.2%). Among the five cases, neurological deficits persisted in two cases and improved in three. The median value of bleeding volume was 112.5 mL (interquartile range, 80.0–200.0). Twenty-five cases (26.0%) needed blood transfusion. The modified Rankin Scale score deteriorated in two cases due to cerebral infarction. The incidence of hemorrhagic and ischemic complications after combined revascularization surgery in patients with ischemic moyamoya disease under antiplatelet therapy was low, indicating the safety of continued antiplatelet therapy.

DOI： 10.18999/nagjms.86.1.82

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DOI： 10.7759/cureus.52524

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Authorship：Last author   Language：English   Publishing type：Research paper (scientific journal)   Publisher：Acta Neurochirurgica  

Background: The use of anchor bolts to secure electrodes to the skull can be difficult in some clinical situations. Herein, we present the boltless technique to secure electrodes to the scalp using nylon sutures to overcome the problems associated with anchor bolts. We investigated the safety, accuracy errors, and patient-related and operative factors affecting errors in the boltless technique. Methods: This single-institution retrospective series analyzed 103 electrodes placed in 12 patients. The target-point localization error (TPLE), entry-point localization error (EPLE), radial error (RE), and depth error (DE) of the electrodes were calculated. Results: The median of the mean operative time per electrode was 9.3 min. The median TPLE, EPLE, RE, and absolute DE value were 4.1 mm, 1.6 mm, 2.7 mm, and 1.9 mm, respectively. Positive correlations were observed between the preoperative scalp thickness, mean operative time per electrode, EPLE, RE, and the absolute value of DE versus TPLE (r =.228, p =.02; r =.678, p =.015; r =.228, p =.02; r =.445, p <.01; r =.630, p <.01, respectively), and electrode approach angle versus EPLE (r =.213, p =.031). Multivariate analysis revealed that the absolute value of DE had the strongest influence on the TPLE, followed by RE and preoperative scalp thickness, respectively (β =.938,.544,.060, respectively, p <.001). No complications related to SEEG insertion and monitoring were encountered. Conclusion: The boltless technique using our unique planning and technical method is a safe, effective, and low-cost alternative in cases where anchor bolts are contraindicated.

DOI： 10.1007/s00701-024-05889-3

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Authorship：Last author   Language：English   Publishing type：Research paper (scientific journal)   Publisher：Cerebral Cortex  

The involvement of the human amygdala in facial mimicry remains a matter of debate. We investigated neural activity in the human amygdala during a task in which an imitation task was separated in time from an observation task involving facial expressions. Neural activity in the amygdala was measured using functional magnetic resonance imaging in 18 healthy individuals and using intracranial electroencephalogram in six medically refractory patients with epilepsy. The results of functional magnetic resonance imaging experiment showed that mimicry of negative and positive expressions activated the amygdala more than mimicry of non-emotional facial movements. In intracranial electroencephalogram experiment and time-frequency analysis, emotion-related activity of the amygdala during mimicry was observed as a significant neural oscillation in the high gamma band range. Furthermore, spectral event analysis of individual trial intracranial electroencephalogram data revealed that sustained oscillation of gamma band activity originated from an increased number and longer duration of neural events in the amygdala. Based on these findings, we conclude that during facial mimicry, visual information of expressions and feedback from facial movements are combined in the amygdalar nuclei. Considering the time difference of information approaching the amygdala, responses to facial movements are likely to modulate rather than initiate affective processing in human participants.

DOI： 10.1093/cercor/bhad488

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DOI： 10.7759/cureus.51850

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BACKGROUND: The surgical techniques for treatment of chronic subdural hematoma (CSDH), a common neurosurgical condition, have been discussed in a lot of clinical literature. However, the recurrence proportion after CSDH surgery remains high, ranging from 10 to 20%. The standard surgical procedure for CSDH involves a craniostomy to evacuate the hematoma, but irrigating the hematoma cavity during the procedure is debatable. The authors hypothesized that the choice of irrigation fluid might be a key factor affecting the outcomes of surgery. This multicenter randomized controlled trial aims to investigate whether intraoperative irrigation using artificial cerebrospinal fluid (ACF) followed by the placement of a subdural drain would yield superior results compared to the placement of a subdural drain alone for CSDH. METHODS: The study will be conducted across 19 neurosurgical departments in Japan. The 1186 eligible patients will be randomly allocated to two groups: irrigation using ACF or not. In either group, a subdural drain is to be placed for at least 12 h postoperatively. Similar to what was done in previous studies, we set the proportion of patients that meet the criteria for ipsilateral reoperation at 7% in the irrigation group and 12% in the non-irrigation group. The primary endpoint is the proportion of patients who meet the criteria for ipsilateral reoperation within 6 months of surgery (clinical worsening of symptoms and increased hematoma on imaging compared with the postoperative state). The secondary endpoints are the proportion of reoperations within 6 months, the proportion being stratified by preoperative hematoma architecture by computed tomography (CT) scan, neurological symptoms, patient condition, mortality at 6 months, complications associated with surgery, length of hospital stay from surgery to discharge, and time of the surgical procedure. DISCUSSION: We present the study protocol for a multicenter randomized controlled trial to investigate our hypothesis that intraoperative irrigation with ACF reduces the recurrence proportion after the removal of chronic subdural hematomas compared with no irrigation. TRIAL REGISTRATION: ClinicalTrials.gov jRCT1041220124. Registered on January 13, 2023.

DOI： 10.1186/s13063-023-07889-7

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OBJECTIVE: Transient neurological events (TNEs) are among the most important events after revascularization surgery in pediatric patients with moyamoya disease (MMD). Although hemodynamic changes and crying are representative factors of TNEs, brain compression by encephalomyosynangiosis (EMS) is another important cause of TNEs. Therefore, the authors assumed that making the EMS as thin as possible reduces the frequency of TNEs. However, thin-split EMS can lead to insufficient development of collateral vessels. This study aimed to evaluate the effects of thin-split EMS in combined revascularization surgery on postoperative outcomes in pediatric patients with MMD. METHODS: The authors retrospectively included 56 consecutive combined revascularization surgeries in the anterior cerebral circulation in pediatric patients with MMD. These surgeries were classified into the former group and thin-split EMS group. The temporal muscle was halved in the former EMS group and split as thinly as possible in the thin-split EMS group. The authors performed between-group comparisons of postoperative stroke events and TNEs during the acute period and the development of collateral flow and stroke events during the chronic period. RESULTS: Former and thin-split EMS procedures were performed in 37 and 19 patients, respectively. TNEs without crying or hemodynamic changes were observed significantly less frequently in the thin-split EMS group than in the former EMS group (0 [0.0%] in the thin-split EMS group vs 9 [24.3%] in the former EMS group, p = 0.021). There were no significant between-group differences in the development of indirect bypass during the chronic period (good: 34 [91.9%] and poor: 3 [8.1%] in the former EMS group vs good: 16 [84.2%] and poor: 3 [15.8%] in the thin-split EMS group; p = 0.397). Additionally, there were no significant between-group differences in the incidence of chronic clinical events, including death or stroke. CONCLUSIONS: Thin-split EMS can reduce TNEs that do not accompany crying or hemodynamic changes. Furthermore, the procedure has sufficient prevention effects against stroke events during the chronic postoperative period in pediatric patients with MMD.

DOI： 10.3171/2023.8.PEDS23295

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OBJECTIVE: This study aimed to analyze whether the development of donor vessels after combined revascularization surgery for moyamoya disease (MMD) is related to cerebral blood flow (CBF) changes. METHODS: We retrospectively reviewed the charts of 11 adult (12 hemispheres) and 13 pediatric (19 hemispheres) patients who underwent combined revascularization in our department. The total vessel cross-sectional area (TVA) was the sum of the cross-sectional areas of the superficial temporal, middle meningeal, and deep temporal arteries imaged using time-of-flight magnetic resonance angiography. The ipsilateral relative CBF (RCBF) on the brain surface in the craniotomy area was calculated by single-photon emission computed tomography. ΔTVA and ΔRCBF were defined as the preoperative and postoperative ratios of TVA and RCBF, and their correlations were analyzed in adult and pediatric patients. RESULTS: The TVA and RCBF showed a significant increase after surgery, regardless of the age group. However, there was no significant correlation between ΔTVA and ΔRCBF in either the adult or pediatric groups. While the adult group exhibited significantly higher ΔRCBF values compared to the pediatric group (p < 0.01, r = -0.44), the ΔTVA values were higher in the pediatric group compared to the adult group (p = 0.06). CONCLUSIONS: In the chronic phase after combined revascularization surgery for MMD, the development of measurable TVA of donor vessels does not necessarily correlate with an increase in CBF around the craniotomy area.

DOI： 10.1016/j.clineuro.2023.108110

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Language：Japanese   Publisher：The Japanese Society of Spinal Surgery  

DOI： 10.2531/spinalsurg.38.71

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Authorship：Last author   Language：English   Publisher：Surgical Neurology International  

Background: Common carotid artery occlusion (CCAO) sometimes requires vascular reconstruction. Ipsilateral superficial temporal artery (STA)-middle cerebral artery (MCA) bypass is unsuitable due to insufficient blood flow to the external carotid artery. The bonnet bypass, one treatment option for CCAO, requires a long coronal incision and bone groove to prevent malposition and collapse of an interposition graft. However, this long incision might lead to skin complications and reduced collateral blood flow. Methods: A 60-year-old man who experienced recurrent ischemic stroke presented with the right internal carotid artery occlusion and left CCAO. The left STA was unavailable; however, both branches of his right STA were well-developed. Minimizing skin invasion was a priority because the patient had diabetes mellitus. We performed a right STA parietal branch – right MCA anastomosis, followed by a right STA frontal branch – left radial artery graft (RAG) – left MCA bonnet bypass using small intermittent skin incisions. Results: We drilled a bone groove extending across the entire length of the interposition graft through the small intermittent skin incisions. Furthermore, we applied a right STA-RAG end-to-side anastomosis instead of an end-to-end anastomosis to preserve collateral skin anastomosis. Postoperatively, the bypass remained patent, and the patient was discharged without complications. Conclusion: The bonnet bypass is a potential treatment for CCAO, but the procedure is invasive. Our modified bonnet bypass method enables less invasive management, preventing collapse and malposition of the interposition graft and minimizing skin complications.

DOI： 10.25259/SNI_528_2024

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Authorship：Last author   Publisher：Journal of Craniovertebral Junction and Spine  

Fibrous dysplasia (FD) is a rare skeletal disorder characterized by the replacement of normal bone with fibrous connective tissue, leading to abnormal bone formation. This case report details the successful treatment of a 61-year-old woman with FD at the craniovertebral junction (CVJ). The patient, who had a history of intracranial meningioma and had already been diagnosed with FD, experienced worsening gait disturbance and muscle weakness following a fall. Imaging studies revealed extensive polyostotic FD lesions in the skull and cervical spine, along with a C2 odontoid fracture causing spinal cord compression. The patient underwent occipitocervical fixation and decompression surgery. Intraoperative O-arm navigation was used to ensure accurate screw placement and effective decompression. This procedure allowed for proper positioning of the C2 and C3 pedicle screws, resection of the hyperplastic occipital bone and C1 posterior arch, and placement of the occipital plate with avoiding the cyst components. At a 2-year follow-up, there were no signs of screw loosening, and the patient showed marked clinical improvement. This case emphasizes the importance of tailored surgical strategies and the use of advanced navigational technologies in managing complex FD cases, particularly those involving the CVJ. It also highlights the challenges of treating polyostotic FD, where complete resection is often unfeasible. The successful outcome in this case supports the use of decompressive surgery combined with stabilization to relieve symptoms and prevent further complications.

DOI： 10.4103/jcvjs.jcvjs_104_24

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Language：Japanese   Publisher：The Japanese Society on Surgery for Cerebral Stroke  

<p>We report a case of low-flow bypass and endovascular coiling for ruptured middle portion posterior communicating artery (PCoA) aneurysm with an ipsilateral internal carotid artery occlusion. A male in his 70s, who presented with headache was referred to our hospital. Brain computed tomography (CT) and CT angiography showed subarachnoid hemorrhage and a cerebral aneurysm in the middle portion of the PCoA, along with an ipsilateral internal carotid artery occlusion. First, we performed a right superficial temporal artery–middle cerebral artery bypass to minimize the risk of perioperative cerebral ischemia and symptomatic vasospasm; subsequently, coil embolization through the posterior circulation was performed. The patient showed a successful recovery at the 3-month follow-up and showed no recurrence 4 years after the treatment. Coil embolization for the middle portion in a PCoA aneurysm has a risk of parent artery occlusion and can result in an incomplete occlusion owing to the small diameter of the PCoA. However, the combination of bypass surgery with coil embolization can be an effective treatment option. This approach reduces the likelihood of perioperative ischemic stroke, symptomatic vasospasm, and recurrence, particularly when the pathogenesis of the aneurysm is hemodynamic stress caused by an occluded artery.</p>

DOI： 10.2335/scs.52.123

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Authorship：Last author   Language：Japanese   Publisher：The Japanese Society of Spinal Surgery  

<p>  Use of drugs and radiological diagnoses are restricted for lumbar disc herniation during pregnancy. Fluoroscopy is avoided as much as possible. We report a case of lumbar disc herniation in a pregnant woman who underwent surgery.</p><p>  A 32-year-old woman, 16 weeks pregnant, who presented to an orthopedic clinic with left lower back and limb pain lasting for a month. Lumbar magnetic resonance imaging showed left posterolateral lumbar disc herniation at L5/S1. Conservative management failed, and she could not tolerate this uncontrollable intense pain until delivery ; therefore, the patient was referred to our department for surgery. She was not paralyzed but was unable to walk because of severe pain. The left straight leg raising test result was <30°. The preoperative Japan Orthopedic Association (JOA) score was 8/29. After consultation with an anesthesiologist and an obstetrician/gynecologist at our hospital, we decided to check the fetal heart sounds and echo findings before and after surgery. Surgery was performed under intravenous anesthesia using the standard Love’s method. The intraoperative position was prone on a four-point trestle table. The vertebral level was checked once, taking care not to irradiate the pelvic cavity. Postoperatively, the sciatica disappeared, the JOA score improved to 29 points, and the patient was discharged without symptoms on the eighth postoperative day.</p><p>  During pregnancy, radiation exposure must be avoided as much as possible, considering its influence on the fetus. As the weeks of pregnancy progress, surgery in the left lateral position is recommended to avoid elevating the intra-abdominal pressure or compressing the inferior vena cava. In our case, the patient’s body shape was almost the same as before she became pregnant. Therefore, we concluded the patient could undergo surgery in the prone position.</p><p>  In cases of herniated discs with severe pain that do not respond to conservative treatment, surgery is extremely useful and safe even during pregnancy.</p>

DOI： 10.2531/spinalsurg.38.61

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Authorship：Last author   Language：English   Publishing type：Research paper (scientific journal)   Publisher：The Japan Neurosurgical Society  

Craniopharyngioma (CP) and Rathke's cleft cyst (RCC) are both suprasellar lesions. They are sometimes difficult to distinguish due to their similar findings. We report a case of papillary craniopharyngioma (pCP) with the clinical findings suggesting RCC. A 42-year-old female with intellectual disability presented to our hospital with severe visual dysfunction. Preoperative images revealed a suprasellar cystic lesion without calcification. We performed transsphenoidal surgery. Since the cyst had condensed-milk-like content suggesting RCC, we performed cyst fenestration and wash without removal of the cyst wall. Thereafter, we found fish-egg-like structures on the cyst wall. The histopathological analysis revealed that they had papillary structures surrounded by hyperplastic squamous epithelium with parakeratosis. Immunostaining for BRAF V600E was positive, leading to the diagnosis of pCP. After the surgery, her visual function improved and follow-up Magnetic resonance imaging at 18 months postoperatively showed no apparent recurrence. The presence of condensed-milk-like content suggests a likelihood of RCC indicating that aggressive resection may not be necessary. In contrast, the existence of fish-egg-like structures suggests pCP and requires careful follow-up.

DOI： 10.2176/jns-nmc.2024-0057

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Authorship：Last author   Language：Japanese   Publisher：Japanese Society for Transplantation and Cellular Therapy  

<p> CD19-redirected chimeric antigen receptor (CAR) T cell therapy has achieved remarkable success in hematological malignancies. However, targeting solid tumor is still challenging. T cell exhaustion is one of the major barriers in tumor microenvironment of solid tumors. In this review, we summarize the multifaceted mechanisms of T cell exhaustion and describe a new insight of CAR T cell exhaustion characterized by a T to NK like transition. Furthermore, the strategies to overcome T cell exhaustion to improve the efficacy of CAR T cell therapy will be addressed.</p>

DOI： 10.7889/tct-23-029

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BACKGROUND: Dermoid cysts located laterally in the posterior fossa are rare. The authors report the case of a dermoid cyst in the cerebellar hemisphere presenting with hemifacial spasm (HFS) caused by multiple vascular attachments due to remote compression effects. OBSERVATIONS: A 48-year-old man presented with left HFS. Computed tomography showed a mass lesion in the left cerebellar hemisphere with calcification and erosion of skull bone. Magnetic resonance imaging showed no contrast enhancement of the lesion and a dural defect. The lesion compressed the brainstem and cerebellopontine cistern, but no vascular attachments to the facial nerve were seen. Tumor removal and microvascular decompression were performed. The lesion was composed of soft tissue containing oil-like liquid and hairs, and the border of the cerebellar arachnoid was clear. There were multiple vascular attachments to the root exit zone, facial nerve, and brainstem. After displacing these arteries, the intraoperative abnormal muscle response disappeared. Histopathological findings showed stratified squamous epithelium, keratin flakes, calcifications, and hairs. The HFS disappeared completely and has remained absent for 27 months. LESSONS: The dermoid cyst originating from occipital bone compressed the cerebellar hemisphere, displacing multiple vessels and leading to HFS. Tumor removal and the removal of all vascular factors can completely resolve HFS.

DOI： 10.3171/CASE23592

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PURPOSE: Artificial intelligence (AI)-based three-dimensional angiography (3D-A) was reported to demonstrate visualization of cerebral vasculature equivalent to that of three-dimensional digital subtraction angiography (3D-DSA). However, the applicability and efficacy of the AI-based 3D‑A algorithm have not yet been investigated for 3D-DSA micro imaging. In this study, we evaluated the usefulness of the AI-based 3D‑A in 3D-DSA micro imaging. MATERIALS AND METHODS: The 3D-DSA micro datasets of 20 consecutive patients with cerebral aneurysm (CA) were reconstructed with 3D-DSA and 3D‑A. Three reviewers compared 3D-DSA and 3D‑A in terms of qualitative parameters (degrees of visualization of CA and the anterior choroidal artery [AChA]) and quantitative parameters (aneurysm diameter, neck diameter, parent vessel diameter, and visible length of AChA). RESULTS: Qualitative evaluation of diagnostic potential revealed that visualization of CA and the proximal to middle parts of the AChA with 3D‑A was equal to that with conventional 3D-DSA; in contrast, visualization of the distal part of the AChA was lower with 3D‑A than with 3D-DSA. Further, regarding quantitative evaluation, the aneurysm diameter, neck diameter, and parent vessel diameter were comparable between 3D‑A and 3D-DSA; in contrast, the visible length of the AChA was lower with 3D‑A than with 3D-DSA. CONCLUSIONS: The AI-based 3D‑A technique is feasible and evaluable visualization of cerebral vasculature with respect to quantitative and qualitative parameters in 3D-DSA micro imaging. However, the 3D‑A technique offers lower visualization of such as the distal portion of the AChA than 3D-DSA.

DOI： 10.1007/s00062-023-01325-8

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Language：English   Publishing type：Research paper (scientific journal)   Publisher：The Japan Neurosurgical Society  

<p>There is a lack of agreement on whether minimally invasive lateral lumbar intervertebral fusion (LLIF) is a suitable treatment option for vertebral fragility fractures (VFFs). Hence, we sought to evaluate the efficacy and safety of LLIF in the management of VFF with neurological deficits in the lumbar spine. Between April 2015 and March 2020, we conducted a retrospective observational study of patients with VFF treated with three-level or less LLIF. The participants had previously received conservative treatment but had not been able to control their neurological symptoms. To assess the outcomes of the LLIF procedures, the patients were followed up for a minimum of 1 year. Clinical and radiological results, which include the timing and location of the bony fusion, were analyzed. The study involved 19 patients with 23 vertebral fracture levels. The residual height of the fractured vertebra was found to be 57.0 ± 12.3% of the height of the adjacent level. The mean Japanese Orthopedic Association score significantly improved postoperatively. Postoperative radiological parameters were significantly maintained at 1 year, and lumbar lordosis was maintained at the last follow-up (45.0 ± 26.7). In total 31 LLIF levels, bone fusion was observed in four levels at 6 months postoperatively, in 16 levels at 1 year, and in 23 levels at the last follow-up. The facet joint had the highest bony fusion location. LLIF within three levels can be safely performed in certain VFF cases with sufficient residual vertebral height.</p>

DOI： 10.2176/jns-nmc.2023-0064

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Optic pathway gliomas (OPGs) are benign tumors that can stop growing or even shrink. In recent years, surgical resection has not been considered the first-line treatment because of its high risk of complications. Chemotherapy is the mainstay of treatment for growing OPGs. Surgical treatment for OPGs with obstructive hydrocephalus is required. Ventriculoperitoneal shunting is effective for all types of hydrocephalus. However, long-term management is required, especially in pediatric cases, and there is a risk of shunt-related complications over a long lifespan. Debulking surgery for OPGs allows us to avoid shunt placement by creating a waterway and releasing the hydrocephalus. To reduce the surgical risk and invasiveness, we used an endoscopic canalization technique with a small-diameter cylinder. In this article, we present a case of endoscopic canalization of an obstructive hydrocephalus caused by OPGs in a 14-year-old female to illustrate our surgical technique.(Trial registration Registry name and number: Efficacy and safety of the neuro-endoscopic treatment for brain tumors (2019-0254)).

DOI： 10.1007/s00381-023-06053-5

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Authorship：Last author   Language：English   Publishing type：Research paper (scientific journal)   Publisher：Springer Science and Business Media LLC  

DOI： 10.1007/s10143-023-02196-w

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Other Link： https://link.springer.com/article/10.1007/s10143-023-02196-w/fulltext.html

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We conducted a study to understand the characteristics of the finishing coils to select the appropriate coil for the final stage of embolization. Consequently, experimental embolization was performed on a 10 mm spherical silicone aneurysm filled with radiolucent coils, which simulated a volume embolization ratio of 20%. Nine different coils (i-ED complex ∞ SilkySoft, SilkySoft, ExtraSoft, V-Trak HyperSoft helical, Barricade 10 complex finishing, Optima complex 10 soft, Target 360 Ultra, Galaxy G3 mini, and Axium prime 3D ExtraSoft) were analyzed six times at random. After each coil insertion, indices that include area, Feret diameter, circularity, and centroid center of mass were calculated using biplane x-ray images. Furthermore, these data were analyzed using the spring constant k, which represents the stiffness of the coil. In multiple comparisons, a significant difference was observed in the area analysis. The i-ED complex ∞ SilkySoft was more widespread than Target 360 Ultra (p < 0.05). However, no significant differences were observed in the other indices. The spring constant k value of Target 360 Ultra was 2.5 times larger than that of the i-ED complex ∞ SilkySoft, and it negatively correlated with the area index rather than with the other indices. Notably, it was suggested that the smaller the spring constant k, the wider the distribution of the finishing coils. Although there was little difference between the coils, some coils had characteristics suggesting that good embolization could be expected using appropriate finishing coils.

DOI： 10.18999/nagjms.85.4.725

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DOI： 10.1016/j.seizure.2023.09.018

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Here, we focused on the association between minor suture fusion and Chiari malformation (CM) occurrence in nonsyndromic craniosynostosis (NSC), and evaluated how the minor suture affects the posterior cranial fossa by measuring the posterior fossa deflection angle (PFA). In this retrospective study, the clinical records of 137 patients who underwent surgery for NSC at Aichi Children's Health and Medical Center between April 2010 and May 2022 were analyzed. Clinical data from Aichi Developmental Disability Center Central Hospital was collected for 23 patients as the external validation set. Among the 137 patients, 123 were diagnosed with NSC and the remaining 14 with syndromic craniosynostosis. Of the 123 NSC patients, 23 patients presented with CM. Multivariate analysis showed that occipito-mastoid fusion was the only significant risk factor for CM (P=0.0218). Within the NSC group, CM patients had a significantly increased PFA (6.33±8.10 deg) compared with those without CM (2.76±3.29 deg, P=0.0487). Nonsyndromic craniosynostosis patients with occipito-mastoid suture fusion had a significantly increased PFA (6.50±7.60 deg) compared with those without occipito-mastoid fusion (2.60±3.23 deg, P=0.0164). In the validation cohort, occipito-mastoid suture fusion was validated as an independent risk factor for CM in univariate analysis. Minor suture fusion may cause CM associated with NSC. Chiari malformation could develop due to an increased PFA due to minor suture fusion, which causes growth disturbance in the affected side and compensatory dilation in the contralateral side within the posterior cranial fossa.

DOI： 10.1097/SCS.0000000000009552

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Authorship：Corresponding author   Language：English   Publishing type：Research paper (scientific journal)   Publisher：Tohoku University Medical Press  

<p>Convection-enhanced delivery (CED) delivers agents directly into tumors and the surrounding parenchyma. Although a promising concept, clinical applications are often hampered by insufficient treatment efficacy. Toward developing an effective CED-based strategy for delivering drugs with proven clinical efficacy, we performed a basic characterization study to explore the locally delivered characteristics of the water soluble nitrosourea nimustine hydrochloride (ACNU). First, ACNU distribution after CED in rodent brain was studied using mass spectrometry imaging. Clearance of ¹⁴C-labeled ACNU after CED in striatum was also studied. ACNU was robustly distributed in rodent brain similar to the distribution of the hydrophilic dye Evans blue after CED, and locally delivered ACNU was observed for over 24 h at the delivery site. Subsequently, to investigate the potential of ACNU to induce an immunostimulative microenvironment, Fas and transforming growth factor-<i>β</i>1 (TGF-<i>β</i>1) was assessed <i>in vitro</i>. We found that ACNU significantly inhibited TGF-<i>β</i>1 secretion and reduced Fas expression. Further, after CED of ACNU in 9L-derived intracranial tumors, the infiltration of CD4/CD8 lymphocytes in tumors was evaluated by immunofluorescence.CED of ACNU in xenografted intracranial tumors induced tumor infiltration of CD4/CD8 lymphocytes. ACNU has a robust distribution in rodent brain by CED, and delayed clearance of the drug was observed at the local infusion site. Further, local delivery of ACNU affects the tumor microenvironment and induces immune cell migration in tumor. These characteristics make ACNU a promising agent for CED.</p>

DOI： 10.1620/tjem.2023.J069

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DOI： 10.1038/s41598-023-45755-3

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Chronic subdural hematoma (CSDH) often causes neurological deterioration and is treated with hematoma evacuation. This study aimed to assess the feasibility of various machine learning models to preoperatively predict the functional outcome of patients with CSDH. Data were retrospectively collected from patients who underwent CSDH surgery at two institutions: one for internal validation and the other for external validation. The poor functional outcome was defined as a modified Rankin scale score of 3–6 upon hospital discharge. The unfavorable outcome was predicted using four machine learning algorithms on an internal held-out cohort (n = 188): logistic regression, support vector machine (SVM), random forest, and light gradient boosting machine. The prediction performance of these models was also validated in an external cohort (n = 99). The area under the curve of the receiver operating characteristic curve (ROC-AUC) of each machine learning-based model was found to be high in both validations (internal: 0.906–0.925, external: 0.833–0.860). In external validation, the SVM model demonstrated the highest ROC-AUC of 0.860 and accuracy of 0.919. This study revealed the potential of machine learning algorithms in predicting unfavorable outcomes at discharge among patients with CSDH undergoing burr hole surgery.

DOI： 10.1038/s41598-023-44029-2

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DOI： 10.1016/j.wneu.2023.07.038

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DOI： 10.1016/j.seizure.2023.07.007

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DOI： 10.1016/j.wneu.2023.07.002

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The occipital transtentorial approach (OTA) is one of the useful approaches to the lesions of the pineal region, dorsal brainstem, and supracerebellar region. However, a wide operative field is sometimes difficult to obtain due to the tentorial sinus and bridging veins. This study evaluated the usefulness of preoperative simulation of OTA, specifically including the cerebellar tentorium in 9 patients. All patients underwent computed tomography angiography and venography and gadolinium-enhanced three-dimensional T1-weighted magnetic resonance images (Gd-3D-T1WI). The images were fused, and the cerebellar tentorium, vessels, and tumor were manually extracted from Gd-3D-T1WI to obtain the simulation images. Visualization of the cerebellar tentorium could discriminate between bridging veins from the occipital lobe and cerebellum, and recognize the site of bridging to the tentorial sinus and variants which may interfere with the tentorial incision. Simulation of the tentorial incision was also possible based on the relationships between the tumor, tentorial sinus, bridging vein, and cerebellar tentorium. The simulation suggested that safe tentorial incision was difficult in two sides because of the crossed tentorial sinus draining the left basal vein and draining veins from the glioblastoma. The OTA was performed in eight cases, and no difficulty was experienced in the tentorial incision in all cases. The simulation findings of the bridging vein and tentorial sinus were consistent with the intraoperative findings. Preoperative simulation including the cerebellar tentorium is useful for determining the optimum and safe side and required extent of the tentorial incision necessary for tumor resection with the OTA.

DOI： 10.1007/s10143-023-02170-6

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BACKGROUND: Spinal cord stimulation (SCS) is one of the neuromodulation therapies for chronic neuropathic pain. The conventional paresthesia-based SCS involves the application of tonic stimulation that induces a sense of paresthesia. Recently, new SCS stimulation patterns without paresthesia have been developed. Differential target multiplexed (DTM) stimulation and fast-acting subperception therapy (FAST) stimulation are the latest paresthesia-free SCS patterns. METHODS: A single-center, open-label, crossover, randomized clinical trial to investigate the superiority of SCS using the latest new stimulation patterns over conventional tonic stimulation for neuropathic pain is planned. This study consists of two steps: SCS trial (first step) and SCS system implantation (second step). In the SCS trial, participants will be randomly assigned to 4 groups receiving stimulation, including tonic, DTM, and FAST. Each stimulation will then be performed for 2 days, and a visual analog scale (VAS) for pain will be evaluated before and after each stimulation pattern. A stimulation-off period for 1 day is set between each stimulation pattern to wash out the residual previous stimulation effects. Pain improvement is defined as more than 33% reduction in the pain VAS. The primary analysis will compare pain improvement between the new stimulation patterns and the conventional tonic stimulation pattern in the SCS trial. The secondary outcomes will be evaluated as follows: (1) the relationships between causative disease and improvement rate by each stimulation pattern; (2) comparison of pain improvement between the DTM and FAST stimulation patterns in all cases and by causative disease; (3) changes in assessment items preoperatively to 24 months after the implantation; (4) preoperative factors associated with long-term effects defined as continuing for more than 12 months; and (5) adverse events related to this study 3 months after the implantation. DISCUSSION: This study aims to clarify the effectiveness of the latest new stimulation patterns compared to the conventional tonic stimulation. In addition, which stimulation pattern is most effective for which kind of causative disease will be clarified. TRIAL REGISTRATION: Japan Registry of Clinical Trials (jRCT) 1,042,220,094. Registered on 21 November 2022, and last modified on 6 January 2023. jRCT is an approved member of the Primary Registry Network of WHO ICTRP.

DOI： 10.1186/s13063-023-07637-x

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DOI： 10.1111/jnp.12345

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Authorship：Lead author,　Last author,　Corresponding author   Language：Japanese   Publishing type：Research paper (scientific journal)   Publisher：株式会社医学書院  

DOI： 10.11477/mf.1436204828

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Language：Japanese   Publisher：(一社)日本神経学会  

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Language：Japanese   Publisher：一般社団法人 日本内分泌学会  

DOI： 10.1507/endocrine.99.s.hpt_4

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Publisher：医歯薬出版  

DOI： 10.32118/ayu28609707

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Oncogene-induced DNA replication stress (RS) and consequent pathogenic R-loop formation are known to impede S phase progression. Nonetheless, cancer cells continuously proliferate under such high-stressed conditions through incompletely understood mechanisms. Here, we report taurine upregulated gene 1 (TUG1) long noncoding RNA (lncRNA), which is highly expressed in many types of cancers, as an important regulator of intrinsic R-loop in cancer cells. Under RS conditions, TUG1 is rapidly upregulated via activation of the ATR-CHK1 signaling pathway, interacts with RPA and DHX9, and engages in resolving R-loops at certain loci, particularly at the CA repeat microsatellite loci. Depletion of TUG1 leads to overabundant R-loops and enhanced RS, leading to substantial inhibition of tumor growth. Our data reveal a role of TUG1 as molecule important for resolving R-loop accumulation in cancer cells and suggest targeting TUG1 as a potent therapeutic approach for cancer treatment.

DOI： 10.1038/s41467-023-40243-8

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BACKGROUND: Dural arteriovenous fistulas (DAVFs) with cortical venous reflux (CVR) carry a high risk for neurological sequelae or death. Recently, transverse-sigmoid sinus DAVFs were shown as good indications for transarterial embolization (TAE) with ONYX. Here, we compared the effectiveness and safety of conventional transvenous embolization (TVE) with those of TAE with ONYX for transverse-sigmoid sinus DAVFs with CVR. METHODS: Sixty-one patients with transverse-sigmoid sinus DAVFs were treated from April 2013 to May 2020. Among them, 37 patients with CVR were included. Transarterial embolization and TVE were compared in terms of complete occlusion and residual CVR immediately after treatment, complications with worsening modified Rankin Scale (mRS) ≥ 1, amount of contrast media used during treatment, radiation exposure, and surgical procedure time. RESULTS: Ten patients were treated with 10 TAEs using ONYX and 27 patients with 29 TVEs. Transarterial embolization and TVE showed no differences in the overall complete occlusion rate (80% [8/10 patients] vs. 80% [23/27], respectively), whereas the residual rate of existing CVR was 10% (1/10 patient) vs. 3.4% (1/27) in the TAE and TVE groups, respectively. No complications with worsening mRS ≥1 occurred in either group. Among the parameters of amount of contrast media, radiation dose, and operative time, only radiation dose in the TAE group was lower than that in the TVE group (median: 2239 mGy vs. 3268 mGy, respectively; p = 0.07). CONCLUSION: For transverse-sigmoid sinus DAVFs with CVR, TAE treatment reduced radiation exposure. However, both TAE and TVE achieved high complete occlusion rates and low complication rates.

DOI： 10.1177/15910199231195135

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Aneurysmal subarachnoid hemorrhage (aSAH) may lead to cerebral vasospasm, significantly associated with morbidity and mortality. In double-blind, placebo-controlled phase 3 studies, clazosentan reduces cerebral vasospasm-related morbidity and all-cause mortality in patients with aSAH. There are no reports about the clinical efficacy of clazosentan combination therapy with some other drugs. Initially, we explored the efficacy of clazosentan combination therapy with cilostazol, statin, and antiepileptic drugs. Subsequently, we assessed the add-on effect of fasudil to clazosentan combination therapy for aSAH patients. This multicenter, retrospective, observational cohort study included Japanese patients with aSAH between June 2022 and March 2023. The primary outcome was the ordinal score on the modified Rankin Scale (mRS; range, 0-6, with elevated scores indicating greater disability) at discharge. Among the 47 cases (women 74.5%; age 64.4 ± 15.0 years) undergoing clazosentan combination therapy, 29 (61.7%) resulted in favorable outcomes. Overall, vasospasm occurred in 16 cases (34.0%), with four cases (8.5%) developing vasospasm-related delayed cerebral ischemia (DCI). Both hypotension and vasospasm-related DCI were related to unfavorable outcome at discharge. Fasudil were added in 18 (38.3%) cases. Despite adding fasudil to clazosentan combination therapy, the incidence of aSAH-related vasospasm did not decrease. Added-on fasudil to combination therapy related to pulmonary edema, vasospasm, and vasospasm-related DCI, and unfavorable outcomes. Clazosentan combination therapy could potentially result in favorable outcomes for aSAH patients to prevent post-aSAH vasospasm-related DCI. The add-on effect of fasudil to combination therapy did not demonstrate a significant impact in reducing aSAH-related vasospasm or improving outcomes at discharge.

DOI： 10.1007/s10143-023-02104-2

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PURPOSE: To examine the specific changes that occur in the expression levels of extracellular vesicle-derived microRNAs (miRNAs) in intracranial cerebrospinal fluid (CSF) in moyamoya disease. METHODS: Patients with arteriosclerotic cerebral ischemia were used as controls to eliminate the effects of cerebral ischemia. Intracranial CSF was collected from moyamoya disease and control patients during bypass surgery. Extracellular vesicles (EVs) were extracted from the CSF. Comprehensive expression analysis of miRNAs extracted from EVs by next-generation sequencing (NGS) and validation by quantitative reverse transcription-polymerase chain reaction (qRT-PCR) was performed. RESULTS: Experiments were conducted on eight cases of moyamoya disease and four control cases. In the comprehensive miRNA expression analysis, 153 miRNAs were upregulated, and 98 miRNAs were downregulated in moyamoya disease compared to the control cases (q-value < 0.05 and |log2 fold change|> 1). qRT-PCR performed on the four most variable miRNAs (hsa-miR-421, hsa-miR-361-5p, hsa-miR-320a, and hsa-miR-29b-3p) associated with vascular lesions among the differentially expressed miRNAs gave the same results as miRNA sequencing. On gene ontology (GO) analysis for the target genes, cytoplasmic stress granule was the most significant GO term. CONCLUSIONS: This study is the first comprehensive expression analysis of EV-derived miRNAs in the CSF of moyamoya disease patients using NGS. The miRNAs identified here may be related to the etiology and pathophysiology of moyamoya disease.

DOI： 10.1007/s00701-023-05579-6

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Authorship：Last author   Language：English   Publishing type：Research paper (scientific journal)   Publisher：The Japan Neurosurgical Society  

Impingement of the common peroneal nerve, a branch of the L5 nerve root, causes common peroneal nerve entrapment neuropathy (CPNE). Although there are cases of CPNE associated with L5 radiculopathy, surgical intervention's effectiveness remains to be elucidated. This retrospective case-control study aimed to evaluate the efficacy of surgery in patients with CPNE associated with L5 radiculopathy. Twenty-two patients (25 limbs) with surgically treated CPNE between 2015 and 2022 were retrospectively reviewed. The limbs were classified into two groups: group R (limbs of CPNE associated with L5 radiculopathy) and group O (limbs of CPNE without L5 radiculopathy). The durations from onset to surgery, the nerve conduction studies (NCSs), and postoperative improvement rates for motor weakness, pain, and dysesthesia were compared between the groups. Group R included 15 limbs (13 patients), and group O included 10 limbs (9 patients). There were no significant differences in the duration from onset to surgery or abnormal findings of NCS between the two groups. The postoperative improvement rates were 88% and 100% (p = 0.62) for muscle weakness, 87% and 80% (p = 0.53) for pain, and 71% and 56% (p = 0.37) for dysesthesia in group R and group O, respectively, without significant differences between groups. CPNE associated with L5 radiculopathy is common, and the results of the present study showed that the surgical outcomes in such cases were satisfactory and comparable to those in CPNE without L5 radiculopathy.

DOI： 10.2176/jns-nmc.2022-0313

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Ependymoma is a rare central nervous system (CNS) tumour occurring in all age groups and is one of the most common paediatric malignant brain tumours. Unlike other malignant brain tumours, ependymomas have few identified point mutations and genetic and epigenetic features. With advances in molecular understanding, the latest 2021 World Health Organization (WHO) classification of CNS tumours divided ependymomas into 10 diagnostic categories based on the histology, molecular information and location; this accurately reflected the prognosis and biology of this tumour. Although maximal surgical resection followed by radiotherapy is considered the standard treatment method, and chemotherapy is considered ineffective, the validation of the role of these treatment modalities continues. Although the rarity and long-term clinical course of ependymoma make designing and conducting prospective clinical trials challenging, knowledge is steadily accumulating and progress is being made. Much of the clinical knowledge obtained from clinical trials to date was based on the previous histology-based WHO classifications, and the addition of new molecular information may lead to more complex treatment strategies. Therefore, this review presents the latest findings on the molecular classification of ependymomas and advances in its treatment.

DOI： 10.1093/jjco/hyad056

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DOI： 10.7759/cureus.42510

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BACKGROUND: Malignant peripheral nerve sheath tumors (MPNSTs) are rare soft-tissue tumors. Intracranial metastasis from MPNSTs is quite rare. OBSERVATIONS: The authors report on a 73-year-old male whose MPNST metastasized to the brain and a 32-year-old male with leptomeningeal metastasis from MPNST and review 41 cases of MPNST that developed intracranial metastasis, as reported in the literature. LESSONS: Brain metastasis and leptomeningeal metastasis of MPNSTs show different clinical courses and require pathology-specific treatment.

DOI： 10.3171/CASE23148

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Language：Japanese   Publisher：Japan Society for Laser Surgery and Medicine  

<p>This study elucidated the relationship between the findings of photodynamic diagnosis (PDD) using 5-aminolevulinic acid (5-ALA) and tumor control in 41 patients undergoing resection of newly diagnosed glioblastoma. All patients underwent maximal resection of contrast-enhanced lesions by PDD using 5-ALA. Intraoperative PDD findings, postoperative lesions with contrast-enhanced residuals, and recurrence sites were analyzed. Residual fluorescent lesion was correlated with the total resection of contrast-enhanced lesion seen in magnetic resonance images. In addition, recurrent disease tended to develop within the lesions with residual fluorescence after resection under white light or blue light. These findings suggested that tumor evaluation using PDD with 5-ALA may aid in the determination of the extent of resection in enhanced lesion and in the prediction of sites for intensification of local treatment.</p>

DOI： 10.2530/jslsm.jslsm-44_0015

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Spheno-orbital meningiomas (SOMs) are complex tumors that grow and extend into nearby structures. SOM tumor growth is often associated with hyperostosis caused by tumor cell infiltration and bone alteration. We describe the case of a 64-year-old man with SOM that extended into the sphenoid sinus without a direct connection between the intracranial and extracranial lesions. This report emphasizes the importance of identifying the growth patterns of SOMs and assessing the paranasal sinuses adjacent to the hyperostotic orbit walls from preoperative images.

DOI： 10.1002/ccr3.7705

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DOI： 10.1161/SVIN.122.000759

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Language：Japanese   Publisher：Movement Disorder Society of Japan (MDSJ)  

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This work aimed to investigate the involvement of the thalamic nuclei in mesial temporal lobe epilepsy (MTLE) and identify the influence of interictal epileptic discharges on the neural basis of memory processing by evaluating the functional connectivity (FC) between the thalamic nuclei and default mode network-related area (DMNRA) using magnetoencephalography. Preoperative datasets of nine patients with MTLE with seizure-free status after surgery and those of nine healthy controls were analyzed. The FC between the thalamic nuclei (anterior nucleus [ANT], mediodorsal nucleus [MD], intralaminar nuclei [IL]), hippocampus, and DMNRA was examined for each of the resting, pre-spike, spike, and post-spike periods in the delta to ripple bands using magnetoencephalography. The FC between the ANT, MD, hippocampus, and medial prefrontal cortex increased in the gamma to ripple bands, whereas the FC between the ANT, IL, and DMNRA decreased in the delta to beta bands, compared with that of the healthy controls at rest. Compared with the rest period, the pre-spike period had significantly decreased FC between the ANT, MD, and DMNRA in the ripple band. Different FC changes between the thalamic nuclei, hippocampus, and DMNRA of specific connections in a particular band may reflect impairment or compensation in the memory processes.

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BACKGROUND: Neuropathic pain after spinal cord injury (SCI), both traumatic and non-traumatic, is refractory to various treatments. Spinal cord stimulation (SCS) is one of the neuromodulation therapies for neuropathic pain, although SCS has insufficient efficacy for neuropathic pain after SCI. The reasons are presumed to be inappropriate locations of SCS leads and conventional tonic stimulation itself does not have a sufficient analgesic effect for the pain. In patients with past spinal surgical histories, the cylinder-type leads are likely to be placed on the caudal side of the SCI because of surgical adhesions. Differential target multiplexed (DTM) stimulation is one of the latest new stimulation patterns that is superior to conventional stimulation. METHODS: A single-center, open-label, randomized, two-way crossover trial is planned to investigate the efficacy of SCS using DTM stimulation placing a paddle lead at the appropriate site for neuropathic pain after SCI in patients with spinal surgical histories. The paddle-type lead delivers energy more efficiently than a cylinder-type lead. This study consists of two steps: SCS trial (first step) and SCS system implantation (second step). The primary outcome is rates of achieving pain improvement with more than 33% reduction 3 months after SCS system implantation. The secondary outcomes are to be evaluated as follows: (1) effectiveness of DTM and tonic stimulations during the SCS trial; (2) changes of assessment items from 1 to 24 months; (3) relationships between the result of the SCS trial and the effects 3 months after SCS system implantation; (4) preoperative factors associated with a long-term effect, defined as continuing for more than 12 months; and (5) whether gait function improves from 1 to 24 months. DISCUSSION: A paddle-type lead placed on the rostral side of SCI and using DTM stimulation may provide significant pain relief for patients with intractable neuropathic pain after SCI in patients with past spinal surgical histories. TRIAL REGISTRATION: Japan Registry of Clinical Trials (jRCT) jRCT 1042220093. Registered on 21 November 2022, and last modified on 6 January 2023. jRCT is approved as a member of the Primary Registry Network of WHO ICTRP.

DOI： 10.1186/s13063-023-07433-7

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The myeloid differentiation primary response gene 88 (MYD88) L265P mutation is a disease-specific mutation of primary central nervous system lymphoma (PCNSL) among the central nervous system tumors. Accordingly, this mutation is considered a reliable diagnostic molecular marker of PCNSL. As the intra-operative diagnosis of PCNSL is sometimes difficult to achieve using histological examinations alone, intra-operative detection of the MYD88 L265P mutation could be effective for the accurate diagnosis of PCNSL. Herein, we aimed to develop a novel rapid genotyping system (GeneSoC) using real-time polymerase chain reaction (PCR) based on microfluidic thermal cycling technology. This real-time PCR system shortened the analysis time, which enabled the detection of the MYD88 L265P mutation within 15 min. Rapid detection of the MYD88 L265P mutation was performed intra-operatively using GeneSoC in 24 consecutive cases with suspected malignant brain tumors, including 10 cases with suspected PCNSL before surgery. The MYD88 L265P mutation was detected in eight cases in which tumors were pathologically diagnosed as PCNSL after the operation, while wild-type MYD88 was detected in 16 cases. Although two of the 16 cases with wild-type MYD88 were pathologically diagnosed as PCNSL after the operation, MYD88 L265P could be detected in all eight PCNSL cases harboring MYD88 L265P. The MYD88 L265P mutation could also be detected using cell-free DNA derived from the cerebrospinal fluid of two PCNSL cases. Detection of the MYD88 L265P mutation using GeneSoC might not only improve the accuracy of intra-operative diagnosis of PCNSL but also help the future pre-operative diagnosis through liquid biopsy of cerebrospinal fluid.

DOI： 10.1111/cas.15762

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OBJECTIVE: This study aimed to evaluate the efficacy and safety of combination therapy with bevacizumab (Bev), irinotecan (CPT-11), and temozolomide (TMZ) in children with central nervous system (CNS) embryonal tumor relapse. METHODS: The authors retrospectively examined 13 consecutive pediatric patients with relapsed or refractory CNS embryonal tumors who received combination therapy comprising Bev, CPT-11, and TMZ. Specifically, 9 patients had medulloblastoma, 3 had atypical teratoid/rhabdoid tumor (AT/RT), and 1 had CNS embryonal tumor with rhabdoid features. Of the 9 medulloblastoma cases, 2 were categorized in the Sonic hedgehog subgroup and 6 in molecular subgroup 3 for medulloblastoma. RESULTS: The complete and partial objective response rates were 66.6% in patients with medulloblastoma and 75.0% in patients with AT/RT or CNS embryonal tumors with rhabdoid features. Furthermore, the 12- and 24-month progression-free survival rates were 69.2% and 51.9% for all patients with recurrent or refractory CNS embryonal tumors, respectively. In contrast, the 12- and 24-month overall survival rates were 67.1% and 58.7%, respectively, for all patients with relapsed or refractory CNS embryonal tumors. The authors observed grade 3 neutropenia, thrombocytopenia, proteinuria, hypertension, diarrhea, and constipation in 23.1%, 7.7%, 23.1%, 7.7%, 7.7%, and 7.7% of patients, respectively. Furthermore, grade 4 neutropenia was observed in 7.1% of patients. Nonhematological adverse effects, such as nausea and constipation, were mild and controlled with standard antiemetics. CONCLUSIONS: This study demonstrated favorable survival outcomes in patients with relapsed or refractory pediatric CNS embryonal tumors and thus helped to investigate the efficacy of combination therapy comprising Bev, CPT-11, and TMZ. Moreover, combination chemotherapy had high objective response rates, and all adverse events were tolerable. To date, data supporting the efficacy and safety of this regimen in the relapsed or refractory AT/RT population are limited. These findings suggest the potential efficacy and safety of combination chemotherapy in patients with relapsed or refractory pediatric CNS embryonal tumors.

DOI： 10.3171/2023.1.PEDS22345

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Authorship：Last author   Language：English   Publishing type：Research paper (scientific journal)   Publisher：The Japan Neurosurgical Society  

The Occipito (O) -C2 angle reflects the correct craniocervical spine alignment; however, the poor image quality of standard intraoperative fluoroscopy at times lead to inaccurate measurements. Herein, we preliminarily investigated the relationship between the O-C2 angle and the Gonion-C2 distance, which is based on the positioning of the mandible and the cervical spine. We enrolled patients who underwent cervical spine radiography in neutral, flexion, and extension positions from January 2020 to October 2020. The difference by posture changes for each parameter was defined as the Δ value, and the Spearman's rank correlation coefficient was determined. Furthermore, we determined the cutoff value of the ΔGonion-C2 distance to predict a decrease of > 10° in the ΔO-C2 angle, which is reported to be related to dysphagia and dyspnea. Seventy-four patients were included. Spearman's rank correlations for the neutral, flexion, and extension positions were 0.630 (P < 0.001), 0.471 (P < 0.001), and 0.625 (P < 0.001), respectively, while the cutoff values of the ΔGonion-C2 distance for predicting > 10° in the ΔO-C2 angle were 9.3 mm for the neutral flexion change (sensitivity: 0.435, specificity: 0.882) and 8.3 mm for the extension-neutral change (sensitivity: 0.712, specificity: 0.909). The O-C2 angle and Gonion-C2 distances correlated; however, this correlation was weaker in the flexed position. Nevertheless, the ΔGonion-C2 distance can be used as a warning sign for postoperative complications after posterior occipital bone fusion surgery, because a decrease of > 10° in the ΔO-C2 angle can be predicted with high specificity.

DOI： 10.2176/jns-nmc.2022-0251

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Stereotactic electroencephalography (SEEG) is receiving increasing attention as a safe and effective technique in the invasive evaluation for epileptogenic zone (EZ) detection. The main clinical question is whether the use of SEEG truly improves outcomes. Herein, we compared outcomes in our patients after three types of intracranial EEG (iEEG): SEEG, the subdural electrode (SDE), and a combined method using depth and strip electrodes. We present here our preliminary results from two demonstrative cases. Several international reports from large epilepsy centers found the following clinical advantages of SEEG: 1) three-dimensional analysis of structures, including bilateral and multilobar structures; 2) low rate of complications; 3) less pneumoencephalopathy and less patient burden during postoperative course, which allows the initiation of video-EEG monitoring immediately after implantation and does not require resection to be performed in the same hospitalization; and 4) a higher rate of good seizure control after resection. In other words, SEEG more accurately identified the EZ than the SDE method. We obtained similar results in our preliminary experiences under limited conditions. In Japan, as of August 2022, dedicated electrodes and SEEG accessories have not been approved and the use of the robot arm is not widespread. The Japanese medical community is hopeful that these issues will soon be resolved and that the experience with SEEG in Japan will align with that of large epilepsy centers internationally.

DOI： 10.2176/jns-nmc.2022-0271

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Invasive brain tumors, especially gliomas, often arise in the eloquent areas of the brain that are involved in language and motor function. The most important goal of brain tumor removal is to safely remove the maximum amount of tumor while preserving neurological function. Maximizing the amount of removed tumor is thought to improve prognosis by prolonging both the progression-free and overall survival periods of patients. In the present study, we review the intraoperative monitoring techniques for motor function-sparing surgery for gliomas arising near the eloquent areas of the brain and electrophysiological monitoring techniques for motor function-sparing surgery for brain tumors arising deep within the brain. In brain tumor surgery, monitoring of direct cortical motor evoked potentials(MEPs), transcranial MEPs, and subcortical MEPs is integral for preserving motor function.

DOI： 10.11477/mf.1436204772

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DOI： 10.2176/jns-nmc.er.2021-0390

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A 19-year-old woman presented with swelling of the left forehead without pain. She did not have any relevant past or family history. Computed tomography showed destruction of the outer cortex of the frontal bone. A solitary mass lesion with a fluid collection was detected with magnetic resonance imaging. Because the swelling of the left forehead had enlarged rapidly with osteolytic changes, surgical removal of the lesion was performed. The lesion appeared to be enveloped in a fibrous capsule. The soft lesion was removed from the frontal bone. The outer frontal bone was absent, although the inner frontal bone was preserved. Then, the frontal bone was resected with margins from the edge of the erosion. The dura mater under the lesion was intact. A cranioplasty was performed using titanium mesh. On histological examination, the trabecular bones revealed irregular shapes and arrangements, indicating fibrous dysplasia. There was a continuous high-cell-concentration pathological lesion outside the fibrous dysplasia. There were numerous cells, such as mononuclear cells, osteoclast-like multinucleated giant cells, foam cells, and red blood cells. The osteoclast-like multinucleated giant cells and other cells did not show significant nuclear atypia. Immunostaining with H3.3G34W was negative, and the ubiquitin-specific peptidase 6/Tre-2 gene showed no rearrangements. The histopathological diagnosis was secondary aneurysmal bone cyst with fibrous dysplasia. Additional postsurgical therapy was not performed. There has been no evidence of recurrence of the lesion for two years.

DOI： 10.18999/nagjms.85.2.395

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Revascularization surgery for young children with moyamoya disease (MMD) is challenging. Although indirect revascularization is preferred because of the technical difficulty in direct anastomosis, higher risks of postoperative infarction remain a problem. We aimed to investigate the effect of superficial temporal artery to middle cerebral artery (STA-MCA) bypass on postoperative outcomes during the acute postoperative period in young children ≤ 5 years old with MMD. This retrospective study included consecutive young children with MMD who underwent surgical revascularization of the anterior cerebral circulation. Groups were determined according to the procedures performed, namely, the combined (STA-MCA bypass with indirect revascularization) and the indirect revascularization groups. The incidences of radiological or symptomatic infarction, transient neurological events, and new neurological deficits that remained at discharge were compared between groups. Of 38 surgical procedures, there were 23 combined and 15 indirect revascularizations. The median age of the patients was 3.0 years, which was significantly different between groups (P < .01). When comparing the postoperative outcomes between groups, the incidences of radiological and symptomatic infarction and new neurological deficits that remained at discharge were significantly lower in the combined revascularization group (P < .05). Logistic regression analysis adjusted for potential confounders found that surgical modality was a statistically significant independent risk factor associated with radiological and symptomatic infarctions (indirect/combined, odds ratio: 10.2; 95% confidence interval: 1.30-79.7; P < .05). STA-MCA bypass combined with indirect revascularization can reduce the incidence of postoperative infarction in young children with MMD and might lead to better neurological outcomes.

DOI： 10.1007/s10143-023-01999-1

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PURPOSE: Delayed hyponatremia (DHN), a unique complication, is the leading cause of unexpected readmission after pituitary surgery. Therefore, this study aimed to develop tools for predicting postoperative DHN in patients undergoing endoscopic transsphenoidal surgery (eTSS) for pituitary neuroendocrine tumors (PitNETs). METHODS: This was a single-center, retrospective study involving 193 patients with PitNETs who underwent eTSS. The objective variable was DHN, defined as serum sodium levels < 135 mmol/L at ≥ 1 time between post operative days 3 and 9. We trained four machine learning models to predict this objective variable using the clinical variables available preoperatively and on the first postoperative day. The clinical variables included patient characteristics, pituitary-related hormone levels, blood test results, radiological findings, and postoperative complications. RESULTS: The random forest (RF) model demonstrated the highest (0.759 ± 0.039) area under the curve of the receiver operating characteristic curve (ROC-AUC), followed by the support vector machine (0.747 ± 0.034), the light gradient boosting machine (LGBM: 0.738 ± 0.026), and the logistic regression (0.710 ± 0.028). The highest accuracy (0.746 ± 0.029) was observed in the LGBM model. The best-performing RF model was based on 24 features, nine of which were clinically available preoperatively. CONCLUSIONS: The proposed machine learning models with pre- and post-resection features predicted DHN after the resection of PitNETs.

DOI： 10.1007/s11102-023-01311-w

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BACKGROUND: In this study, we propose a butterfly needle tap and suction (BTS) technique for recurrent chronic subdural hematoma (CSDH) as an alternative to reoperation with burr hole craniostomy (BHC) and investigate its efficacy and safety. The procedure involves percutaneous puncture through the burr hole created during the previous surgery and subsequent hematoma evacuation using a butterfly needle. METHODS: This retrospective study included patients who underwent BTS for CSDH at Ogaki Municipal Hospital between January 2017 and December 2020. The follow-up CT scans were reviewed after several weeks. We evaluated the number of percutaneous punctures required to resolve CSDH during the BTS technique, the volume of the evacuated hematoma, and procedure-related complications. RESULTS: Twenty-six patients were enrolled in the study, 21 of whom achieved resolution of the hematoma using punctures with the BTS technique alone (mean, 2.2 ± 1.5). Five patients had a recurrence of hematoma after one or more punctures during the BTS technique, and they underwent reoperation with BHC according to the surgeon's decision or patient requests. Among the 55 punctures, 43.0 ± 16.0 ml of hematoma was evacuated per puncture. The evacuated hematoma volume was 41.9 ± 16.4 ml in the BTS-alone group and 49.4 ± 12.9 ml in the reoperation group, with no significant difference (p = 0.25). Three patients complained of a headache during the puncture procedure, and no other complications, including intracranial hemorrhage or infection, were reported therein. CONCLUSIONS: The BTS technique is an effective alternative to reoperation with BHC.

DOI： 10.1007/s00701-023-05543-4

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The 2021 World Health Organization (WHO) classification of central nervous system tumors applied molecular criteria and further integrated histological and molecular diagnosis of gliomas. This classification allows for the diagnosis of isocitrate dehydrogenase wild-type (IDHwt) glioblastoma (GBM), and WHO grade 4 with histologically lower-grade gliomas (LrGGs), even in the absence of high-grade histopathologic features, such as necrosis and/or microvascular proliferation. They contain at least one of the following molecular features: epidermal growth factor receptor amplification, chromosome 7 gain/10 loss, or telomerase reverse transcriptase promoter mutation. In the imaging features at the time of histological diagnosis, a gliomatosis cerebri growth pattern was frequently observed in these tumors. Furthermore, this growth pattern was significantly higher in IDHwt GBM, WHO grade 4, with histological grade II gliomas. Although the exact prognosis of IDHwt GBM, WHO grade 4, with histologically LGGs remains unknown, its OS was approximately 1-2 years similar to that of histologically IDHwt GBM, WHO grade 4, despite histopathological features similar to IDHmut LrGGs. These findings reinforce the need for the analysis of molecular features, regardless of presenting similar clinical characteristics and imaging features to IDHmut LrGGs.

DOI： 10.1007/s10014-023-00458-5

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Adult-type diffuse gliomas are divided into Astrocytoma, IDH-mutant, Oligodendroglioma, IDH-mutant and 1p/19q-codeleted and Glioblastoma, IDH-wildtype based on the IDH mutation, and 1p/19q codeletion status. To determine the treatment strategy for these tumors, pre-operative prediction of IDH mutation and 1p/19q codeletion status might be effective. Computer-aided diagnosis (CADx) systems using machine learning have been noted as innovative diagnostic methods. However, it is difficult to promote the clinical application of machine learning systems at each institute because the support of various specialists is essential. In this study, we established an easy-to-use computer-aided diagnosis system using Microsoft Azure Machine Learning Studio (MAMLS) to predict these statuses. We constructed an analysis model using 258 adult-type diffuse glioma cases from The Cancer Genome Atlas (TCGA) cohort. Using MRI T2-weighted images, the overall accuracy, sensitivity, and specificity for the prediction of IDH mutation and 1p/19q codeletion were 86.9%, 80.9%, and 92.0%, and 94.7%, 94.1%, and 95.1%, respectively. We also constructed an reliable analysis model for the prediction of IDH mutation and 1p/19q codeletion using an independent Nagoya cohort including 202 cases. These analysis models were established within 30 min. This easy-to-use CADx system might be useful for the clinical application of CADx in various institutes.

DOI： 10.1007/s10014-023-00459-4

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The goal of this study is to perform correlation analysis of Computed tomography (CT) and magnetic resonance imaging (MRI) results in posterior ligament complex (PLC) injury and define the morphological traits of thoracolumbar (TL) burst fractures connected to PLC injury. Forty patients with surgically repaired TL burst fractures between January 2013 and December 2020 were retrospectively analyzed. The patients were split into two groups for comparison based on MRI (Group P: patients with a confirmed or suspected PLC injury; Group N: patients with PLC injury denied). The radiographic morphological examination based on CT scans and clinical evaluation was performed and compared between two groups. The thoracolumbar injury classification and severity score (TLICS), the load sharing classification (LSC) scores, and the number of patients with neurological impairments were considerably greater in Group P. Loss of height of the fracture (loss height), local kyphosis of the fracture (local kyphosis), and supraspinous distance were significantly higher in Group P and significantly associated with PLC injuries indicating severe vertebral body destruction and traumatic kyphosis in multivariate logistic analysis [odds ratio: 1.90, 1.06, and 1.13, respectively]. Cutoff value for local kyphosis obtained from the receiver operating characteristic curve was 18.8. If local kyphosis is greater than 18.8 degrees on CT scans, we should take into account the probability of the highly damaged burst fracture associated with PLC injury. In this situation, we should carefully assess MRI to identify the spinal cord injury or spinal cord compression in addition to PLC injury because these instances likely present with neurological abnormalities.

DOI： 10.2176/jns-nmc.2021-0390

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BACKGROUND: The pathophysiology of neurovascular compression syndrome has not been fully elucidated, and cerebrospinal fluid levels of nerve tissue-related markers involved in this disorder have not yet been reported. METHODS: We measured cerebrospinal fluid levels of S100B protein, neuron-specific enolase, and myelin basic protein in 21 patients with trigeminal neuralgia, 9 patients with hemifacial spasms, and 10 patients with non-ruptured intracranial aneurysms (control). Cerebrospinal fluid levels of these markers were determined using commercially available assay kits. RESULTS: Both trigeminal neuralgia and hemifacial spasm groups showed significantly increased cerebrospinal fluid levels of S100B compared with the control group (1120 [IQR 391-1420], 766 [IQR 583-1500], and 255 [IQR 190-285] pg/mL, respectively; p = 0.001). There were no statistically significant differences in cerebrospinal fluid levels of neuron-specific enolase or myelin basic protein among the groups. CONCLUSION: Cerebrospinal fluid S100B levels were significantly higher in patients with trigeminal neuralgia and hemifacial spasm than in controls, which suggests the involvement of S100B in the underlying pathophysiology of neurovascular compression syndrome.

DOI： 10.1007/s00701-022-05434-0

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Abstract

This study investigated the effectiveness and safety of low-dose salvage craniospinal irradiation (CSI) for recurrent germinoma. We retrospectively reviewed long-term tumor control and late adverse effects in 15 recurrent germinoma patients treated at our hospital between 1983 and 2019. Following the first recurrence of germinoma, seven were treated with 24–30 Gy of salvage CSI, three underwent non-CSI, and five were treated with only chemotherapy. CSI achieved a significantly better recurrence-free survival rate after the first recurrence compared to other strategies (100% vs 33%, p &amp;lt; 0.001: log-rank test). To evaluate the safety of salvage CSI, we assessed the outcomes at the final follow-up of seven patients who received salvage CSI at first recurrence and three patients who received salvage CSI at second recurrence. The median follow-up period was 220 months after initial treatment. Five patients who received 40–50 Gy of radiation therapy or underwent multiple radiation therapy before salvage CSI were classified into Group A, whereas five patients treated with platinum-based chemotherapy and 24–32 Gy of radiation therapy to the primary site, whole ventricle, or whole brain were classified into Group B. In Group A, one had endocrine dysfunction and the other had visual dysfunction. None were socially independent. Meanwhile, in Group B, no endocrine or visual dysfunction was found, and three patients were socially independent. Salvage CSI achieved excellent tumor control in recurrent germinoma and was safe in patients initially treated with low-dose radiation therapy and chemotherapy.

DOI： 10.1093/jrr/rrac095

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BACKGROUND: Developmental venous anomalies (DVAs) are congenital anatomical variants of the normal deep parenchymal veins. DVAs are occasionally found incidentally on brain imaging, and most cases are asymptomatic. However, they rarely cause central nervous disorders. Herein, a case of mesencephalic DVA that caused aqueduct stenosis and hydrocephalus and discuss its diagnosis and treatment is reported. OBSERVATIONS: The patient was a 48-year-old female who presented with depression. Computed tomography and magnetic resonance imaging (MRI) of the head revealed obstructive hydrocephalus. Contrast-enhanced MRI revealed an abnormally distended linear region with enhancement on the top of the cerebral aqueduct, which was confirmed as a DVA by digital subtraction angiography. An endoscopic third ventriculostomy (ETV) was performed to improve the patient's symptoms. Intraoperative endoscopic imaging showed obstruction of the cerebral aqueduct by the DVA. LESSONS: This report describes a rare case of obstructive hydrocephalus caused by DVA. It highlights the usefulness of contrast-enhanced MRI for diagnosing cerebral aqueduct obstructions due to DVAs and the effectiveness of ETV as a treatment option.

DOI： 10.3171/CASE22563

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INTRODUCTION: The pituitary gland, regulating various hormones, is central in the endocrine system. As spontaneous recovery from hypopituitarism is rare, and exogenous-hormone substitution is clumsy, pituitary replacement via regenerative medicine, using pluripotent stem cells, is desirable. We have developed a differentiation method that in mice yields pituitary organoids (POs) derived from human embryonic stem cells (hESC). Efficacy of these POs, transplanted subcutaneously into hypopituitary mice, in reversing hypopituitarism was studied. METHODS: hESC-derived POs were transplanted into inguinal subcutaneous white adipose tissue (ISWAT) and beneath dorsal skin, a relatively avascular region (AR), of hypophysectomized severe combined immunodeficient (SCID) mice. Pituitary function was evaluated thereafter for ¾ 6mo, assaying basal plasma ACTH and ACTH response to corticotropin-releasing hormone (CRH) stimulation. Histopathologic examination of organoids 150d after transplantation assessed engraftment. Some mice received an inhibitor of vascular endothelial growth factor (VEGF) to permit assessment of how angiogenesis contributed to subcutaneous engraftment. RESULTS: During follow-up, both basal and CRH-stimulated plasma ACTH levels were significantly higher in the ISWAT group (p < 0.001 - 0.05 and 0.001 - 0.005, respectively) than in a sham-operated group. ACTH secretion also was higher in the ISWAT group than in the AR group. Histopathologic study found ACTH-producing human pituitary-cell clusters in both groups of allografts, which had acquired a microvasculature. POs qPCR showed expression of angiogenetic factors. Plasma ACTH levels decreased with VEGF-inhibitor administration. CONCLUSIONS: Subcutaneous transplantation of hESC-derived POs into hypopituitary SCID mice efficaciously renders recipients ACTH-sufficient.

DOI： 10.3389/fendo.2023.1130465

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OBJECTIVE: Moyamoya disease (MMD) is a rare cerebrovascular disease characterized by progressive stenosis of the internal carotid artery (ICA) and secondary formation of collateral vessels. Revascularization surgery is performed in patients with MMD to prevent stroke; however, the pathogenesis of MMD remains unknown. Recently, long noncoding RNAs (lncRNAs) have been found to play a key role in gene regulation and are implicated in various vascular diseases. However, the lncRNA expression profile in MMD lesions has not been investigated. In this study the authors aimed to determine the characteristics of lncRNA expression in MMD lesions. METHODS: The authors collected microsamples of the middle cerebral artery (MCA) from patients with MMD (n = 21) and patients with control conditions (n = 11) who underwent neurosurgical treatment. Using microarray experiments, the authors compared the profiles of lncRNA expression in the MCAs of the MMD and control patient groups and identified differentially expressed lncRNAs (fold change > 2, q < 0.05). In addition, the neighboring coding genes, whose transcription can be regulated in cis by the identified differentially expressed lncRNAs, were investigated and Gene Ontology (GO) analysis was applied to predict associated biological functions. RESULTS: The authors detected 308 differentially expressed lncRNAs (fold change > 2, q < 0.05), including 306 upregulated and 2 downregulated lncRNAs in the MCA from patients with MMD. Regarding the prediction of biological function, GO analyses with possible coding genes whose transcription was regulated in cis by the identified differentially expressed lncRNAs suggested involvement in the antibacterial humoral response, T-cell receptor signaling pathway, positive regulation of cytokine production, and branching involved in blood vessel morphogenesis. CONCLUSIONS: The profile of lncRNA expression in MMD lesions was different from that in the normal cerebral artery, and differentially expressed lncRNAs were identified. This study provides new insights into the pathophysiology of MMD.

DOI： 10.3171/2022.5.JNS22579

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OBJECTIVE: The protocol for antiplatelet therapy after stent-assisted coil embolization (SACE) for intracranial aneurysms is not well established. In particular, the indications for single antiplatelet therapy (SAPT) discontinuation remain controversial. The authors investigated the long-term outcomes of SAPT discontinuation after SACE among patients at a single institution. METHODS: Patients who underwent SACE during the period from 2010 to 2020 and who were followed up for > 1 year were included in this study. The delayed ischemic and hemorrhagic complication rates were examined during follow-up. Moreover, the risk factors of antiplatelet therapy reduction or discontinuation and the outcomes of SAPT discontinuation were examined. RESULTS: In total, 240 patients were included in the analysis. The average patient age was 60.3 years, and the average follow-up period was 46.7 months. Nine (3.8%) patients presented with symptomatic delayed ischemic complication, and 3 (1.3%) patients experienced a decline in modified Rankin Scale score. The stent configuration (T- or Y-stent) was the only risk factor associated with delayed ischemic complication (p < 0.001). SAPT was discontinued in 147 (71.7%) of 205 patients who were followed up for > 2 years, and no ischemic complications were observed. CONCLUSIONS: It is safe to discontinue SAPT in patients without ischemic complications and with stable intraaneurysmal signals on MRA 2 years after SACE. The T- or Y-stent is a high-risk factor for delayed ischemic complications, and antiplatelet therapy reduction or discontinuation should be cautiously considered.

DOI： 10.3171/2022.6.JNS22815

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PURPOSE: Postoperative pituitary dysfunction, a critical problem in the treatment of craniopharyngiomas, can occur even when the pituitary stalk is preserved. We hypothesized that compromise of the primary superior hypophyseal artery (pSHA) might be related to this occurrence. METHODS: We performed a retrospective review of 131 patients with craniopharyngioma who underwent surgery from April 2009 to September 2021. The inclusion criteria were initial surgery, endoscopic transsphenoidal surgery, preoperative normal pituitary function or pituitary dysfunction in one axis, and morphological preservation of the pituitary stalk. The branches of the pSHA consist mainly of the chiasmatic branches (Cb), infundibular branches (Ib), and descending branches (Db). We analyzed the association between postoperative pituitary function and preservation of these branches. RESULTS: Twenty patients met the criteria. Preoperative anterior pituitary function was normal in 18 patients, and there was isolated growth hormone deficiency in two patients. No patient had preoperative diabetes insipidus (DI). Anterior pituitary function was unchanged postoperatively in eight patients. Of these eight patients, bilateral preservation of pSHA Ib was confirmed in seven patients. Bilateral preservation of pSHA Ib was the only factor associated with preserved anterior pituitary function (p < 0.01). Fifteen patients were free of permanent DI, and the preservation of any given pSHA branch produced no significant difference in the postoperative occurrence of permanent DI. CONCLUSIONS: Our study shows that bilateral preservation of pSHA Ib provides favorable postoperative anterior pituitary function in craniopharyngioma surgery; however, such preservation may have little effect on the postoperative occurrence of DI.

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BACKGROUND: Rituximab, high-dose methotrexate (HD-MTX), procarbazine and vincristine (R-MPV), has significantly prolonged the survival of patients with primary central nervous system lymphoma (PCNSL), but predictive factors for response to R-MPV have not yet been investigated. Herein, we investigated the correlation of MYD88 L265P and CD79B Y196 mutations, which are the most frequently found molecular alterations in PCNSL, with prognosis of patients with PCNSL treated with R-MPV. METHODS: We investigated the long-term clinical course and status of MYD88 and CD79B genes in 85 patients with PCNSL treated with R-MPV or HD-MTX treatment, and the correlation of these genetic mutations with prognosis. RESULTS: R-MPV achieved an excellent tumor control rate (61.6% and 69.9% of 5-year progression-free and overall survival rates, respectively). While MYD88 L265P mutation had no significant effect on survival, patients with CD79B Y196 mutations exhibited prolonged survival (p < 0.05). However, the association of CD79B Y196 mutation with a better prognosis was not observed in the HD-MTX cohort, which indicated that CD79B Y196 mutation was a predictive marker for a favorable response to R-MPV. Furthermore, we established an all-in-one rapid genotyping system for these genetic mutations. CONCLUSIONS: In conclusion, CD79B Y196 mutation is a potent predictive marker for favorable response to R-MPV in PCNSL. The rapid identification of MYD88 L265P and CD79B Y196 mutations can be helpful not only for the accurate molecular diagnosis of PCNSL but also for the prediction of response to R-MPV.

DOI： 10.1002/cam4.5512

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OBJECTIVE: To study the compared surgical and radiographic outcomes of Transvertebral foraminotomy (TVF) with anterior cervical discectomy and fusion (ACDF) in patients with unilateral cervical spondylotic radiculopathy (CSR). METHODS: We performed a retrospective comparative study of 72 consecutive patients with 1- or 2-level CSR treated with ACDF or TVF. 27 patients who underwent TVF (group T) and 45 patients who underwent ACDF (group A) with a minimum 2-year follow-up were enrolled. We evaluated clinical outcomes and radiological assessment. Clinical outcome included Visual analog scale (VAS) scores for axial, arm pain at preoperatively and final follow-up. VAS score for painful swallowing was also evaluated 1 week after surgery. Radiological assessment included C2-7 sagittal Cobb angle (C2-7 CA), range of motion (ROM) of C2-7 CA, the height, angle and ROM of the functional spinal unit (FSU), and tip of the spinous process of the operated segment. We also evaluated the disc height, FSU angle, and ROM of the FSU at the cranial adjacent segment. RESULTS: Both groups had good clinical outcomes. Soft tissue swelling was significantly less prominent in group T than that for group A. VAS scores for painful swallowing is lower in group T without significant difference. The ROM of C2-7 CA, FSU, and spinous processes demonstrated a significant reduction in group A compared with group T.(P < 0.05). Disc height at the cranial adjacent segment was maintained in group T. CONCLUSIONS: TVF is as effective as ACDF for unilateral CSR and preserves whole cervical spine and segmental alignment.

DOI： 10.1016/j.wneu.2022.12.053

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A 57-year-old man had trigeminal schwannoma in Meckel's cave with eyelid myokymia only. The evaluation of the trigeminal-evoked blink reflex was useful for confirming eyelid myokymia and contributed to surgical decision-making. In patients with persistent eyelid myokymia, neurophysiological and imaging evaluations may be useful for determining the underlying pathophysiology.

DOI： 10.1002/ccr3.7086

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Language：Japanese   Publisher：(一社)日本神経学会  

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BACKGROUND: Approximately 70% of lower-grade gliomas harbor isocitrate dehydrogenase 1 (IDH1) mutations, resulting in accumulation of oncometabolite D-2-hydroxyglutarate (D-2-HG); this leads to epigenetic dysregulation, oncogenesis, and subsequent clonal expansion. DS-1001 is an oral brain-penetrant mutant IDH1 selective inhibitor. This first-in-human study investigated the safety, pharmacokinetics, pharmacodynamics, and efficacy of DS-1001. METHODS: This was a multicenter, open-label, dose-escalation, phase I study of DS-1001 for recurrent/progressive IDH1-mutant (R132) glioma (N = 47) (NCT03030066). DS-1001 was administered orally at 125-1400 mg twice daily. Dose escalation used a modified continual reassessment method. RESULTS: The maximum tolerated dose was not reached. Eight patients were continuing treatment at the data cut-off. Most adverse events (AEs) were grade 1-2. Twenty patients (42.6%) experienced at least one grade 3 AE. No grade 4 or 5 AEs or serious drug-related AEs were reported. Common AEs (>20%) were skin hyperpigmentation, diarrhea, pruritus, alopecia, arthralgia, nausea, headache, rash, and dry skin. The objective response rates were 17.1% for enhancing tumors and 33.3% for non-enhancing tumors. Median progression-free survival was 10.4 months (95% confidence interval [CI], 6.1 to 17.7 months) and not reached (95% CI, 24.1 to not reached) for the enhancing and non-enhancing glioma cohorts, respectively. Seven on-treatment brain tumor samples showed a significantly lower amount of D-2-HG compared with pre-study archived samples. CONCLUSIONS: DS-1001 was well-tolerated with a favorable brain distribution. Recurrent/progressive IDH1-mutant glioma patients responded to treatment. A study of DS-1001 in patients with chemotherapy- and radiotherapy-naïve IDH1-mutated WHO grade 2 glioma is ongoing (NCT04458272).

DOI： 10.1093/neuonc/noac155

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BACKGROUND: Pineal parenchymal tumors of intermediate differentiation (PPTIDs) are rare in the pineal gland. A case of PPTID that disseminated to the lumbosacral spine 13 years after the total resection of a primary intracranial tumor has been reported. OBSERVATIONS: A 14-year-old female presented with headache and diplopia. Magnetic resonance imaging revealed a pineal tumor that induced obstructive hydrocephalus. A biopsy and endoscopic third ventriculostomy were performed. Histological diagnosis revealed a grade II PPTID. Two months later, the tumor was removed via craniotomy because the postoperative Gamma Knife surgery was ineffective. Histological diagnosis confirmed PPTID, although the grade was revised from II to III. Postoperative adjuvant therapy was not performed, because the lesion had been irradiated and gross total tumor removal was achieved. She has had no recurrence in 13 years. However, pain around the anus newly appeared. Magnetic resonance imaging of the spine revealed a solid lesion in the lumbosacral spine. The lesion was subtotally resected, and histological diagnosis revealed grade III PPTID. Postoperative radiotherapy was performed, and she had no recurrence 1 year after radiotherapy. LESSONS: Remote dissemination of PPTID can occur several years after the initial resection. Regular follow-up imaging, including the spinal region, should be encouraged.

DOI： 10.3171/CASE22475

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Glioblastoma sometimes develops with acute onset due to intracerebral hemorrhage. Although it is sometimes difficult to diagnose patients with hemorrhagic-onset glioblastoma at the acute phase of intracerebral hemorrhage (ICH), the progressive enlargement of perifocal edema or the development of contrast-enhanced lesion triggers the diagnosis of glioblastoma within six months. Herein, we present a rare case of glioblastoma in which the diagnosis was delayed as long as 17 months after ICH. A 62-year-old man presented with a headache and aphasia. Computed tomography revealed ICH in the left temporal lobe. Magnetic resonance (MR) images revealed that the hematoma had a mix of isointense and surrounding hypointense lesions on T1-weighted MR images and gadolinium-enhanced lesions at the wall and the septum of the hematoma. An endoscopic evacuation of the hematoma was performed. No causative lesions were found during intraoperative and histological examinations. After seven months, abnormal signals were completely resolved on MR images, except for the small and stable enhanced lesion on three-dimensional gadolinium-enhanced T1-weighted MR imaging (3D Gd-T1WI) at the base of the hematoma, which did not change in size for seven months. However, a large gadolinium-enhanced lesion at the left temporal lobe developed 17 months after ICH. He underwent total resection of the lesion and was diagnosed with glioblastoma. He received radiation therapy and temozolomide but died of disseminated recurrence 31 months after ICH. In conclusion, this report presents a didactic case of glioblastoma in which the diagnosis of glioblastoma was delayed 17 months after ICH whereas hemorrhagic-onset glioblastoma was previously considered ruled out in cases in which six months or more have passed after ICH. In order not to overlook these cases, follow-up with 3D Gd-T1WI is essential in the case of suspected tumor-related ICH and close follow-up is recommended when the enhanced lesion does not resolve after a long period even if it does not grow.

DOI： 10.7759/cureus.34672

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Digital subtraction angiography (DSA) is the most useful technique for diagnosing spinal arteriovenous malformations (AVM). In recent years, with the improvement of imaging capabilities, the usefulness of three-dimensional (3D) imaging by fusing various modalities has been recognized. The use of 3D fusion imaging with a workstation connected to an angiography system has been reported in many cases of intracranial disease, but less frequently for spinal AVM. In this article, we describe two illustrative cases of spinal AVM in which 3D fusion imaging was useful for treatment. Although 3D fusion images using the system have the disadvantage that only a maximum of two images can be fused, it provides spinal surgeons with useful information for preoperative evaluation in a small amount of time.

DOI： 10.18999/nagjms.85.1.127

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OBJECTIVE: To evaluate the static and dynamic features of blood blister-like aneurysms (BBAs) using cerebral angiography to identify characteristic features to improve the diagnosis of these uncommon aneurysms. METHODS: Digital subtraction angiography (DSA) images were compared between patients with BBAs (n = 12, group A) and patients with unruptured paraclinoid aneurysms ≤ 5 mm in size treated by endovascular procedures (n = 12, group B). DSA images were assessed for irregularities in the diameter of the parent artery and delayed inflow and outflow of contrast medium in the aneurysm. Enlargement of the aneurysm and morphological changes from the first assessment were also evaluated in patients with BBAs. RESULTS: Compared to the group B, group A had a higher proportion of irregular vessel diameter (p = 0.013) and the delayed contrast medium outflow (p = 0.014). As well, stagnation of contrast medium along the aneurysm wall was a characteristic finding of BBAs, even for small aneurysms. CONCLUSION: Irregular morphological features of the parent artery and delayed contrast medium outflow as characteristic findings of ruptured BBAs may improve the diagnosis of these uncommon aneurysms, which remains challenging in practice.

DOI： 10.1016/j.clineuro.2022.107583

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In the neuroendovascular field, the training of operators has become an important issue. Recently, eye-tracking technology has been introduced into various fields of medical education. This study aimed to apply eye-tracking technology to the training of neuroendovascular therapy. Six neurosurgeons, including three neuroendovascular specialists and three trainees, at our institution and related facilities participated in the study. Eye movement was recorded by the eye-tracking device during the microcatheter navigation and coil placement into the silastic aneurysm model under biplane X-ray fluoroscopy. Eye-tracking analysis during neuroendovascular therapy was feasible in all six subjects. In microcatheter navigation, specialists tended to more frequently switch their attention between frontal and lateral images than trainees. In coil embolization, the overall gaze frequency tended to increase, and the average fixation duration tended to decrease as the number of experienced cases increased. Inexperienced operators tend to fix their gaze when they are operators than when they are assistants. More experienced operators tended to look at the microcatheter longer in the coil insertion task. The eye-tracking analysis may be useful for operator training in neuroendovascular therapy. Experts may have moved their eyes more frequently than trainees to gaze at the right place. In the future, it will be necessary to collect gaze data for more operators in various tasks.

DOI： 10.18999/nagjms.85.1.50

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BACKGROUND: T2-fluid-attenuated inversion recovery (FLAIR) mismatch sign is a specific imaging finding of isocitrate dehydrogenase (IDH)-mutant astrocytomas. Histologically, a hypointense area on FLAIR images indicates the presence of microcysts. Here we report a case of IDH-mutant astrocytoma that shrunk spontaneously. CASE DESCRIPTION: A 26-year-old woman presented with a complaint of headache. Her magnetic resonance (MR) images revealed a lesion mass with a T2-FLAIR mismatch sign in the left frontal lobe. Subsequently, after 1 month, she was referred to our department, and we found that the lesion had unexpectedly shrunk; however, no further shrinkage was observed in the next 3 months. Furthermore, a biopsy was performed, and the results indicated a diagnosis of astrocytoma, IDH-mutant CNS WHO grade 3. Thus, she underwent subtotal resection. We found no neurological deficits in the patient, and she received 60 Gy of radiotherapy at the local site and chemotherapy with nimustine hydrochloride (ACNU), followed by the administration of ACNU every 8 weeks for 2 years. Overall, after 36 months of tumour resection, she was in good health and exhibited no recurrence. Notably, her histological and MR image findings suggested that the macroscopic cyst was formed by the fusion of microcysts, which is a characteristic feature of IDH-mutant astrocytoma with a T2-FLAIR mismatch sign, and that the tumour shrunk because of the rupture of the cyst in the Sylvian cistern. CONCLUSION: The present case report suggests that IDH-mutant astrocytoma cannot be ruled out even when the lesion shrinks spontaneously.

DOI： 10.1080/02688697.2023.2170328

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OBJECTIVE: An autologous formalin-fixed tumor vaccine (AFTV) derived from resected glioblastoma (GBM) tissue can be used against unidentified tumor antigens. Thus, the authors conducted a multicenter double-blind phase IIb trial to investigate the efficacy of an AFTV. METHODS: Eligible patients were adults with supratentorial GBMs, 16-75 years of age, with Karnofsky Performance Scale (KPS) scores ≥ 60%, and no long-term steroid administration. An AFTV comprising fixed paraffin-embedded tumor tissue with immune adjuvants or an identical placebo without fixed tumor tissue was injected intradermally over three courses before and after chemoradiotherapy. The primary and secondary end points were overall survival (OS), progression-free survival (PFS), and 3-year survival rate. RESULTS: Sixty-three patients were enrolled. The average patient age was 61 years. The median KPS score was 80%, and the median resection rate was 95%. The full analysis set of 57 patients indicated no significant difference in OS (p = 0.64) for the AFTV group (median OS 25.6 months, 3-year OS rate 38%) compared with the placebo group (31.5 months and 41%, respectively) and no difference in PFS (median PFS 13.3 months in both groups, p = 0.98). For patients with imaging-based total tumor removal, the 3-year PFS rate was 81% in the AFTV group versus 46% in the placebo group (p = 0.067), whereas the 3-year OS rate was 80% versus 54% (p = 0.16), respectively. Similar results were obtained in the p53-negative subgroups. Severe adverse effects were not observed. CONCLUSIONS: The AFTV may have potential effects in certain patient subgroups. A phase III study for patients with total tumor removal remains warranted to confirm these findings. Clinical trial registration no.: UMIN000010602 (UMIN Clinical Trials Registry).

DOI： 10.3171/2022.12.JNS221221

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Isocitrate dehydrogenase wild-type (IDHwt) diffuse astrocytomas feature highly infiltrative patterns, such as a gliomatosis cerebri growth pattern with widespread involvement. Among these tumors, localized IDHwt histologically diffuse astrocytomas are rarer than the infiltrative type. The aim of this study was to assess and describe the clinical, radiographic, histopathological, and molecular characteristics of this rare type of IDHwt histologically diffuse astrocytomas and thereby provide more information on how its features affect clinical prognoses and outcomes. We retrospectively analyzed the records of five patients with localized IDHwt histologically diffuse astrocytomas between July 2017 and January 2020. All patients were female, and their mean age at the time of the initial treatment was 55.0 years. All patients had focal disease that did not include gliomatosis cerebri or multifocal disease. All patients received a histopathological diagnosis of diffuse astrocytomas at the time of the initial treatment. For recurrent tumors, second surgeries were performed at a mean of 12.4 months after the initial surgery. A histopathological diagnosis of glioblastoma was made in four patients and one of gliosarcoma in one patient. The initial status of IDH1, IDH2, H3F3A, HIST1H3B, and BRAF was "wild-type" in all patients. TERT promoter mutations (C250T or C228T) were detected in four patients. No tumors harbored a 1p/19q codeletion, EGFR amplification, or chromosome 7 gain/10 loss (+ 7/ - 10). We assessed clinical cases of localized IDHwt histologically diffuse astrocytomas that resulted in malignant recurrence and a poor clinical prognosis similar to that of glioblastomas. Our case series suggests that even in patients with histologically diffuse astrocytomas and those who present with radiographic imaging findings suggestive of a localized tumor mass, physicians should consider the possibility of IDHwt histologically diffuse astrocytomas.

DOI： 10.1038/s41598-022-25928-2

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Cerebrovascular complications of central nervous system tuberculosis (TB) are predictors of poor prognosis and adverse outcomes. These complications are mainly intracranial arterial involvement, with occasional venous involvement. Here, we present a 67-year-old woman with concurrent cerebral infarction and intracranial tuberculoma induced by the carotid plaque complicated by miliary tuberculosis. Mycobacterium tuberculosis was observed on the luminal side of the carotid plaques in pathological specimens. Treatment with anti-TB drugs alone would likely not cure the patient, as M. tuberculosis would continue to disseminate. Endarterectomy could directly remove the embolic source, and a complete cure was achieved.

DOI： 10.1007/s00701-023-05489-7

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OBJECTIVE: The efficacy of magnetic resonance-guided focused ultrasound (MRgFUS) ablation for essential tremor (ET) is well known; however, no prognostic factors have been established. The authors aimed to retrospectively investigate MRgFUS ablation outcomes and associated factors and to define the cutoff values for each prognostic factor. METHODS: Sixty-four Japanese patients who underwent unilateral ventral intermediate nucleus thalamotomy with MRgFUS for ET were included. Follow-up evaluations were performed at 1 week and 1, 3, 6, 12, and 24 months postoperatively. Tremor suppression was evaluated using the Clinical Rating Scale for Tremor (CRST), and adverse effects were recorded postoperatively. Outcome-associated factors were examined preoperatively, intraoperatively, and postoperatively using multivariate analyses. The cutoff values for the prognostic factors were calculated using receiver operating characteristics. RESULTS: Percentage improvements in the CRST scores of the affected upper limb were 82.4%, 72.2%, 68.6%, and 65.9% at 1, 3, 6, and 12 months, respectively. Preoperatively, a high skull density ratio (SDR) (p ≤ 0.047), low CRST part B score (used to assess tremors during several tasks) (cutoff value 25, p ≤ 0.041), and nonoccurrence of resting tremors (p = 0.027) were significantly associated with improved tremor control. An intraoperatively high maximum mean temperature (cutoff value 52.5°C, p ≤ 0.047), postoperatively large lesion (cutoff value 3.9 mm in the anterior-posterior direction, p ≤ 0.002; cutoff value 5.0-5.55 mm in the superior-inferior direction, p ≤ 0.026), and small transducer focus correction (p ≤ 0.015) were also associated with improved tremor control. No valid cutoff value was found for SDR. Adverse effects (limb weakness, sensory disturbance, ataxia/walking disturbance, dysgeusia, dysarthria, and facial swelling) occurred transiently and were associated with high SDR, high temperature, high number of sonication sessions, large lesion, and occurrence of resting tremor. Patients who developed leg weakness experienced greater percentage improvement in tremors at 3 months postoperatively than those who did not. CONCLUSIONS: MRgFUS ablation could be used to achieve good tremor control with acceptable adverse effects in Japanese patients with ET. The relatively low SDR in Asian ethnic groups as compared with that of Western populations makes treatment difficult; however, the cutoff values obtained in this study may be useful for achieving good treatment outcomes even in such patients. Clinical trial registration no.: UMIN000026952 (University Hospital Medical Information Network). ABBREVIATIONS: ACPC = anterior commissure-posterior commissure; AP = anterior to posterior; CRST = Clinical Rating Scale for Tremor; ET = essential tremor; MRgFUS = magnetic resonance-guided focused ultrasound; PC = posterior commissure; PSA = posterior subthalamic area; RL = right to left; ROC = receiver operating characteristic; SDR = skull density ratio; SI = superior to inferior; T2WI = T2-weighted imaging; VIM = ventral intermediate nucleus.

DOI： 10.3171/2022.3.JNS212460

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Understanding the semiology and underlying anatomy of each seizure is essential for epilepsy surgeons. According to the International League Against Epilepsy(ILAE)classification in 2017, seizure types are classified as focal, generalized, or unknown onset, all of which are further classified as motor or non-motor onset. Impairment of awareness is involved in consciousness systems(consisting of subcortical structures such as the thalamus and upper brain stem)and cortical structures(including the frontoparietal association cortices). Seizures with motor features are divided into elementary symptoms for which myoclonic, clonic, and tonic expressions reflect the somatotopy of the primary motor cortex; and integrated or gestural motor expression representing activation of the motor association cortex. A rostrocaudal gradient is demonstrated in hyperkinetic movements in frontal lobe epilepsy. Non-motor epileptic features should be understood together with auras, which correspond to focal aware seizures and hold crucial localizing semiologic values. The correlation between functional anatomy and seizure semiology is justified by invasive recordings such as stereotactic electroencephalography and subdural recordings, and also confirmed by seizure outcomes after resection of supposed epileptogenic zones. In addition to the conventional localization theory, it is necessary to consider the neural network theory for further recognition of the functionally anatomical basis in an incomprehensible demonstration of seizures.

DOI： 10.11477/mf.1436204711

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Language：Japanese   Publisher：(一社)日本定位・機能神経外科学会  

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Language：Japanese   Publisher：The Japanese Society for Pediatric Neurosurgery  

<p>The effect of surgical resection on recurrent pediatric malignant brain tumors is still controversial. We analyzed cases of seven patients who underwent surgical resection of recurrent lesions.</p><p>In all patients, the Karnofsky performance status was not aggravated after multiple surgeries. The progression free survival periods in four patients with posterior infratentorial ependymomas were 10, 18, 24, and 35 months. Three of the four patients survived about 100 months following multiple repeat surgeries and re-radiation. However, one patient died because of disseminated lesions 21 months after the initial therapy. These results suggested that repeat surgeries for recurrent ependymomas were effective unless disseminated lesions were detected. However, the effects of repeat surgeries were modest in our patients with glioblastoma. Based on the World Health Organization’s Classification of Central Nervous System tumors, 5^th Edition, these tumors were diagnosed as diffuse astrocytoma, <i>IDH</i>-mutant, grade 4, and diffuse hemispheric tumor, <i>H3 G34</i>-mutant. Although these tumors did not recur as leptomeningeal dissemination, local tumor control was very poor by repeat surgeries. Finally, one infant case with <i>SHH</i>, <i>TP53</i>-wild type medulloblastoma survived more than 100 months following repeat surgeries and chemotherapy alone. The indication of repeat surgeries for medulloblastoma might be limited; thus, precise molecular and histological information was useful to determine therapy.</p>

DOI： 10.34544/jspn.48.1_33

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<p>  Malignant glioma is one of the most intractable malignant neoplasms. Standard treatment consists of surgery, radiotherapy, and chemotherapy, although substantial therapeutic effects cannot be expected. In particular, the blood-brain barrier (BBB) protects the central nervous system, reducing the effectiveness of chemotherapy. Temozolomide is the standard treatment, but it only prolongs the median survival time by about two months compared to surgery and radiation. Molecular-targeted therapies, which have revolutionized the treatment of many cancer types, have all failed to treat malignant gliomas, and the focus is now on the spatial and temporal diversity of tumors. In addition, the problem of drug penetration across the BBB has been refocused. Precision medicine is also attracting great expectations, but panel testing can only be done once. Even if an expert panel recommends a treatment, there is a problem in that most treatments have to rely on investigational therapies. It is also known that even if there is a draggable genetic abnormality in tumors, drugs targeting those abnormalities are often ineffective against brain tumors. Therefore, it is necessary that all novel therapeutic approaches, such as virus therapy, neutron capture therapy, and drug delivery techniques like convection-enhanced delivery, be employed to improve treatment outcomes in malignant glioma.</p>

DOI： 10.7887/jcns.32.148

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<p><b>Objective</b>: We verified the usefulness of patient management using a balloon-pressurized belt (Stanch Belt Plus) to prevent puncture site hematomas, which can occur at a specific rate even with hemostatic devices after endovascular neurosurgery.</p><p><b>Methods</b>: A total of 113 patients who underwent endovascular surgery with a femoral puncture from April 2019 to September 2020 were divided into two groups: 31 cases using a traditional compression belt and 82 cases using a newly introduced balloon-pressurized belt during this period. The clinical data were analyzed retrospectively. The chi-square test and Mann–Whitney U test were used to test for significant differences.</p><p><b>Results</b>: There were no significant differences in treatment procedures or frequency of hemostatic device use, but the balloon-pressurized belt group had a significantly lower incidence of hematomas (2.4% vs 12.9%, p <0.05) and a significantly lower incidence of moderate or higher lumbago (22.0% vs 41.9%, p <0.05). The incidence of epidermal detachment tended to be low; however, no significant difference was observed (3.7% vs. 12.9%, n.s.).</p><p><b>Conclusion</b>: Patient management with the newly introduced balloon-pressurized belt may decrease the occurrence of groin hematoma and lumbago among complications after endovascular neurosurgery.</p>

DOI： 10.5797/jnet.oa.2023-0053

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DOI： 10.1016/j.abst.2023.09.001

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<p>In this article, we report a case wherein a brain tumor was suspected based on computed tomography and magnetic resonance imaging findings. We made an initial diagnosis of malignant brain tumor based on methionine-positron emission tomography (PET) findings, but the correct diagnosis was dural arteriovenous fistula (DAVF). The patient was a 45-year-old man with DAVF who developed headache. Methionine-PET imaging showed high methionine uptake in the lesion. Although the tumor was strongly suspected from the findings of methionine-PET, the diagnosis of DAVF could be made correctly only by interpreting digital subtraction angiography and computed tomographic angiography. The findings of methionine-PET, which is considered useful in the diagnosis and denial of brain tumors, made the diagnosis of DAVF more difficult. The increased uptake of methionine-PET in DAVF is an important finding because, to our knowledge, this study is the first to report such finding. The results of this study might be useful for differential diagnoses when the diagnosis is uncertain.</p>

DOI： 10.2176/jns-nmc.2022-0055

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Language：English   Publisher：The Japan Neurosurgical Society  

<p>Choroid plexus carcinomas (CPCs) are rare malignant tumors of neuro-ectodermal origin, accounting for less than 1% of all intracranial tumors. The recurrence rates of CPCs are very high and typically occur in the short-term following surgery, even after gross total removal. Here we present a rare case of CPC with spinal metastasis, which occurred long after its initial presentation. A 25-year-old woman with a history of increased intracranial pressure underwent resection for a tumor of the fourth ventricle, with a histopathological diagnosis of CPC. After tumor resection, she received 30 Gy of radiation therapy to the craniospinal axis and 20 Gy to the primary site, followed by nimustine hydrochloride chemotherapy. The residual lesion completely responded to these treatments. She suffered sensory loss in the sacral region 13 years later, followed by refractory skin ulcer in the sacral region 17 years after the initial treatments. Magnetic resonance imaging at 17 years after the initial treatments showed tumor in the sacral region, which was enlarged upon follow-up after 18 months, causing incontinence and loss of urinary intention. She underwent tumor resection, with a histological diagnosis of recurrent CPC. She received salvage re-irradiation. This case shows that CPC can spread via the cerebrospinal fluid pathways and cause spinal metastasis, with relatively slow clinical course. The present case suggests that patients with CPCs may need long-term follow-up imaging of the total neural axis to identify late recurrence at both the primary site and spinal metastasis.</p>

DOI： 10.2176/jns-nmc.2022-0056

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<p>Arachnoiditis ossificans (AO) is a rare disease, wherein ossified lesions in the subarachnoid space obstruct the flow of spinal fluid or compress the spinal cord, thereby causing myelopathy. Here we describe a rare case of AO and discuss the diagnosis and treatment strategies for this disease. A 66-year-old man with a history of subarachnoid hemorrhage presented with gait disturbance and dysuria for 7 months. Spinal magnetic resonance imaging and computed tomography (CT) myelography showed syringomyelia at the T5-T8 level and dorsally tethered spinal cord at the T8-T10 level. Preoperative noncontrast CT was not performed. The patient was diagnosed with adhesive arachnoiditis and underwent arachnoidolysis. However, intraoperative findings showed the presence of ossification lesions on the dorsal surface of the spinal cord, and intraoperative ultrasound (IOU) showed a hyperintense lesion with acoustic shadowing on the dorsal surface of the spinal cord, with limited visibility of the spinal cord. After removal of the lesions, IOU showed untethered and well-decompressed spinal cord and restoration of cerebrospinal fluid pulsation. Based on these findings, the patient was finally diagnosed with AO, which is an extremely rare disease, with an unknown frequency of occurrence. Therefore, all patients with adhesive spinal arachnoiditis require a preoperative noncontrast CT scan to evaluate for ossification lesions. In this case, we were fortunate to be able to treat AO with IOU, which demonstrated specific findings.</p>

DOI： 10.2176/jns-nmc.2022-0036

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BACKGROUND: Two-dimensional digital subtraction angiography (2D-DSA) and conventional three-dimensional digital subtraction angiography (3D-DSA) are used for the detailed analysis of dural arteriovenous fistula (DAVF). Recently, four-dimensional digital subtraction angiography (4D-DSA), a novel technology, has been attracting attention. The current study aimed to evaluate the capability of 4D-DSA in assessing anatomical angioarchitecture in DAVF. METHODS: In total, 10 consecutive patients with DAVF who underwent 3D-DSA and 4D-DSA at a single institution were included in the analysis. Initially, one-slice multiplanar reconstruction (MPR) images obtained via 4D-DSA and 3D-DSA were compared to investigate the visibility of the feeding artery, fistulous point, and draining vein. Next, 4D-DSA images alone were compared and evaluated with and the MPR images of conventional 3D-DSA in terms of diagnosis of the angioarchitecture. RESULTS: In total, six men and four women (with a mean age of 65.6 ± 10.0 years) were included in the study. The MPR image obtained via 3D-DSA had a significantly better visibility of the feeding artery and fistulous point than that acquired via 4D-DSA (p < 0.05). As for the draining vein, the score was equivalent and not significant. The diagnosis of the vascular architecture of only 4D-DSA images was nearly equivalent to that of MPR images of 3D-DSA. There were no inter-rater differences. CONCLUSION: The MPR images obtained via 4D-DSA may be slightly inferior to those acquired via 3D-DSA in identifying fine angioarchitecture in DAVF. However, they were comparable in terms of diagnostic accuracy.

DOI： 10.1177/15910199221145526

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Holmes tremor is a symptomatic tremor that develops secondary to central nervous system disorders. Stereotactic neuromodulation is considered when the tremors are intractable. Targeting the ventral intermediate nucleus (Vim) is common; however, the outcome is often unsatisfactory, and the posterior subthalamic area (PSA) is expected as alternative target. In this study, we report the case of a patient with intractable Holmes tremor who underwent dual-lead deep brain stimulation (DBS) to stimulate multiple locations in the PSA and thalamus. The patient was a 77-year-old female who complained of severe tremor in her left upper extremity that developed one year after her right thalamic infarction. Vim-thalamotomy using focused ultrasound therapy (FUS) was initially performed but failed to control tremor. Subsequently, we performed DBS using two leads to stimulate four different structures. Accordingly, one lead was implanted with the aim of targeting the ventral oralis nucleus (Vo)/zona incerta (Zi), and the other with the aim of targeting the Vim/prelemniscal radiation (Raprl). Electrode stimulation revealed that Raprl and Zi had obvious effects. Postoperatively, the patient achieved good tremor control without any side effects, which was maintained for two years. Considering that she demonstrated resting, postural, and intention/action tremor, and Vim-thalamotomy by FUS was insufficient for tremor control, complicated pathogenesis was presumed in her symptoms including both the cerebellothalamic and the pallidothalamic pathways. Using the dual-lead DBS technique, we have more choices to adjust the stimulation at multiple sites, where different functional networks are connected. Intractable tremors, such as Holmes tremor, may have complicated pathology, therefore, modulating multiple pathological networks is necessary. We suggest that the dual-lead DBS (Vo/Raprl and Vim/Zi) presented here is safe, technically feasible, and possibly effective for the control of Holmes tremor.

DOI： 10.3389/fnhum.2022.1065459

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We performed virological analysis of resected brain tissues from a patient with temporal lobe epilepsy associated with mesial temporal sclerosis after febrile status epilepticus caused by human herpesvirus 6 infection. The patient had febrile status epilepticus at 9 months of age associated with human herpesvirus 6 infection. Magnetic resonance imaging revealed reduced water diffusion in the right temporal lobe and hippocampus. Polymerase chain reaction analysis detected 1.6 × 105 copies/μg of human herpesvirus 6 DNA in whole blood, but none in the cerebrospinal fluid. The patient developed temporal lobe epilepsy associated with mesial temporal sclerosis at 67 months of age, necessitating surgical treatment. Anterior temporal lobectomy was performed at 171 months of age. Real-time polymerase chain reaction analysis of resected brain tissues revealed no viral DNA. In our patient, human herpesvirus 6 infection triggered febrile status epilepticus, while direct evidence to prove contribution of HHV-6 to the development of MTS was not obtained.

DOI： 10.1002/epi4.12634

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Language：Japanese   Publisher：(一社)日本脊髄外科学会  

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Other Link： https://search.jamas.or.jp/default/link?pub_year=2022&ichushi_jid=J02943&link_issn=&doc_id=20230112220009&doc_link_id=10.2531%2Fspinalsurg.36.296&url=https%3A%2F%2Fdoi.org%2F10.2531%2Fspinalsurg.36.296&type=J-STAGE&icon=https%3A%2F%2Fjk04.jamas.or.jp%2Ficon%2F00007_3.gif

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In a 30-year-old woman with cervical ependymoma, magnetic resonance imaging (MRI) features changed over a short period of time. A T2-weighted MRI scan obtained 1 month before surgery showed an intramedullary tumor with mixed intensity suggesting solid and cystic components at the C4-7 level. Gadolinium-enhanced MRI showed ring enhancement, including a cystic component at the C4-7 level. However, fluorodeoxyglucose positron emission tomography revealed uptake at the C5 level, and another gadolinium-enhanced MRI scan performed 24 days after the previous scan showed only enhanced lesions corresponding to the solid component at the C5-6 level. These images changed over a short time because the spontaneous hematoma, which was enhanced on the first MRI scan, had disappeared later. Fluorodeoxyglucose positron emission tomography or repeated gadolinium-enhanced MRI may be useful if the initial MRI scan suggests the presence of a hematoma in spinal ependymoma.

DOI： 10.1016/J.WNEU.2022.08.116

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OBJECTIVE: The biopsy procedure is intended to obtain an adequate specimen volume from the targeted area while ensuring minimal damage to the normal brain. We performed navigation-guided endoscopic biopsy using a small-diameter cylinder to reduce the invasiveness of the biopsy procedure and ensure a sufficient amount of tissue is collected. We examined whether it is possible to reduce brain tissue injury by using a small-diameter cylinder and improve safety and effectiveness by using an endoscope to directly observe the lesion and achieve hemostasis. METHODS: Patients who underwent endoscopic biopsy surgery using a 6-mm-diameter cylinder for intraparenchymal lesions were enrolled in this study. Postoperative hematoma formation and the extent of trajectory scarring were assessed. RESULTS: Fifty-two procedures performed on 51 patients were analyzed in this study. Postoperative neurological deterioration was not observed in any patient. A pathological diagnosis was made for all patients. Postoperative computed tomography revealed no hematoma after 49 procedures and a small hematoma after 3 procedures, and no patients required additional treatment. A postoperative trajectory scar less than 5 mm in diameter was observed after 30 procedures, a scar of 5-10 mm was observed after 19 procedures, a scar larger than 10 mm was observed after 3 procedures at 1 week after surgery, and 40, 6 and 0 scars were observed at 3 months after surgery. CONCLUSIONS: Endoscopic biopsy using a small-diameter cylinder is a possible alternative biopsy technique for intraparenchymal lesions. This surgical technique is useful, especially in patients at risk of hemorrhagic complications.

DOI： 10.1016/j.wneu.2022.08.147

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Thromboembolic complications are a concern in the treatment of cerebral aneurysms using a flow diverter. In this study, we report a case of abnormal foreshortening of a Flow Re-Direction Endoluminal Device (FRED) caused by in-stent thrombosis immediately after its deployment. A 72-year-old woman had a large cavernous carotid aneurysm, which caused ptosis and diplopia. FRED deployment was planned, and dual antiplatelet therapy was initiated 2 weeks before the procedure. Under systemic heparinization, FRED was deployed with local compaction over the aneurysm orifice. Cone-beam computed tomography subsequently revealed slightly poor wall apposition at the proximal side. While the balloon catheter was prepared for angioplasty, the stent became abnormally foreshortened, the proximal side slipped into the aneurysm, and the internal carotid artery became occluded. FRED was removed using a snare wire, and recanalization was obtained. The lumen of the removed FRED was filled with thrombus. The antiplatelet therapy was changed to triple regimen, and a Pipeline Flex embolization device was placed 1 month later. At that time, no thromboembolic complications were noted. It was considered that thrombotic occlusion was followed by foreshortening of FRED on the distal side because of antegrade blood flow. Multiple factors, such as increased mesh density by locally compacted stent deployment, slightly poor wall apposition, clopidogrel resistance, and the dual-layer structure of FRED, may have been involved in thrombus formation.

DOI： 10.18999/nagjms.84.4.884

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This study sought to establish an experimental aneurysm model of visualizing coil insertion using radiolucent nylon coils. Moreover, this study aimed to clarify the characteristics and differences of each coil and use them clinically as indices of coil selection. The coil insertion test was performed on the 10 mm spherical silicone aneurysm model filled to a nylon coil volume embolization ratio of 11.8%. Five types of coil were randomly tested six times, and the distribution of the coils was analyzed by fluoroscopy imaging. Indices of "Area (mm2)," "Feret's diameter (mm)," and "Circularity" were calculated from the fluoroscopic images. Among the indices, only "Area" showed a significant difference between coils (p = 0.002). On multivariate analysis, "Area" of the ED Infini was larger than those of Target XL soft and Galaxy G3 (p = 0.018 and 0.026, respectively). Furthermore, the area of the 360 soft was larger than that of G3 (p = 0.049). Analysis of the correlation between these values and the coil configuration showed that "Area" was negatively correlated with the stock-wire diameter (r = -0.50; p = 0.004) and primary coil configuration (r = -0.65; p < 0.001). When inserting the coils in the early stage, although the difference between each coil is relatively difficult to obtain, knowledge on the proper use of the coils with differences in characteristics can help in selecting the coil most appropriate for the conditions.

DOI： 10.18999/nagjms.84.4.762

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Autosomal dominant polycystic kidney disease (ADPKD) is a rare hereditary disease leading to end-stage renal failure in approximately half of patients by seventy years of age. It is important to continuously take tolvaptan to control disease progression. However, adherence to tolvaptan in a real-world setting, rather than randomized controlled trials (RCTs), has not been sufficiently reported. We aimed to investigate tolvaptan persistence among patients with ADPKD using a large claims database. Using the Shizuoka Kokuho Database, we identified patients diagnosed with ADPKD who were prescribed tolvaptan from March 2014-September 2018 in Japan. The persistence rate of tolvaptan medication was estimated by Kaplan-Meier analysis, and patient background factors associated with treatment discontinuation were exploratively evaluated with log-rank tests. We identified 1714 eligible patients with ADPKD, and among them, 25 patients used tolvaptan medication. We followed up these patients, whose median treatment duration was 21 months. The persistence rates at 12, 24, and 36 months were estimated to be 70.8% (95% confidence interval: 48.2-93.4), 46.5% (23.2-66.9), and 38.7% (16.4-60.8), respectively. In the exploratory analysis, there were no factors that were obviously associated with tolvaptan discontinuation. The persistence rate of tolvaptan in patients with ADPKD in a real-world setting may be lower than that in previous RCTs. Our innovative method, particularly in Japan, to analyze adherence using large claims data should change the way clinical epidemiological research and health policies of rare diseases are designed in the future.

DOI： 10.1097/MD.0000000000030923

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Radiation therapy is a well-established, minimally invasive method of treating brain tumors. In recent years, the number of post-radiotherapy patients has increased, and delayed side effects are evident. De novo aneurysm formation after radiation often manifests as fatal subarachnoid hemorrhage (SAH), resulting in severe clinical outcomes. Nevertheless, the prognosis and therapeutic efficacy of radiation-induced aneurysms (RIAs) remain unclear. Using the PubMed database from 1980 to 2021, we screened 45 articles (53 individual cases) on RIAs; approximately 70% of RIAs were diagnosed after rupture. Of 38 ruptured RIAs, 12 (31.6%) had modified Rankin scale (mRS) 5-6. On the other hand, all unruptured RIAs (15 cases) recovered without neurological deficits (p = 0.012). Ten of the 39 ruptured RIAs were treated surgically, and 22 were treated endovascularly. There was no significant difference in mRS between treatment modalities (p = 0.393), but conservative therapy was significantly related to unfavorable outcomes (p = 0.025). To improve clinical outcomes, RIAs need to be diagnosed before rupture. Surgeons should be aware of de novo aneurysm formation in patients long after radiation therapy.

DOI： 10.1007/s10143-022-01820-5

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The evidence that heart rate variability (HRV) decreases during early Parkinson's disease (PD) largely depends on electrocardiogram data. In this study, we examined HRV in PD using wearable sensors and assessed various evaluation methods for detecting disease-related alterations. We evaluated 27 patients with PD and 23 disease controls. The wearable sensors POLAR V800 HR and POLAR H10 were used for the HRV measurements. The participants wore the two sensors for approximately 24 h, and long-term HRV data were acquired. We analyzed the standard deviation of normal R-R intervals (SDNN) and coefficient of variation of R-R intervals (CVRR) for every 100 consecutive beats. Focusing on the fluctuation of SDNN and CVRR, we extracted the minimum, first decile, first quartile, and median values of SDNN and CVRR. The area under the receiver operating characteristic curve (AUC) for each HRV parameter was calculated to differentiate PD from the disease controls. The minimum values of SDNN and CVRR had the highest AUC (SDNN: AUC 0.90, 95% confidence interval [CI] 0.78-0.96; CVRR: AUC 0.90, CI 0.76-0.96) among the evaluation methods tested. The minimum values of SDNN and CVRR were significantly decreased in PD (SDNN: 9.5 ± 4.0 ms vs. 4.4 ± 2.0 ms, p < 0.0001; CVRR: 1.15 ± 0.33% vs. 0.65 ± 0.24%, p < 0.0001). We detected decreased HRV in PD using wearable sensors. Analyzing the minimum values of the HRV parameter in long-term recordings appears to be appropriate for detecting the decrease in HRV in PD.

DOI： 10.1007/s00702-022-02528-y

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OBJECTIVE: Cerebral cavernous malformations (CMs) presenting with focal neurological symptoms or mass effects require surgical removal. In recent years, cylindrical retractors have been widely utilized for the removal of deep-seated lesions during both microscopic and endoscopic surgery. In the present study, we evaluated the efficacy and safety of endoscopic transcylinder removal of CMs using a novel wet-field technique. METHODS: We included 13 patients with supratentorial CMs who had undergone endoscopic transcylinder surgery between April 2013 and March 2022. One patient experienced recurrence of the CM and underwent a second endoscopic transcylinder surgery. Therefore, we retrospectively evaluated 14 procedures. The surgical field was continuously irrigated with artificial cerebrospinal fluid to maintain expansion and visualization of the tumor bed. We termed this method as the "wet-field technique." Patient characteristics, symptoms, and pre- and postoperative magnetic resonance imaging results were obtained from medical records. RESULTS: The average maximum CM diameter was 35.3 mm (range: 10-65 mm). Cylinder diameters were 6 mm in eight procedures, 10 mm in four procedures, and 17 mm in one procedure. Wet-field technique was applied in all cases. The endoscope provided a bright field of view even under water. Continuous water irrigation made it easier to observe the entire tumor bed which naturally expanded by water pressure. Gross total resection was achieved in 13 procedures, while subtotal resection was achieved in one procedure. No surgical complications were observed. CONCLUSIONS: The endoscopic transcylinder removal using wet-field technique is safe and effective for the removal of symptomatic intracranial supratentorial CMs.

DOI： 10.1007/s00701-022-05273-z

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BACKGROUND AND PURPOSE: West syndrome is a developmental and epileptic encephalopathy characterized by epileptic spasms, neurodevelopmental regression, and a specific EEG pattern called hypsarrhythmia. Our aim was to investigate the brain activities related to hypsarrhythmia at onset and focal epileptiform discharges in the remote period in children with West syndrome using simultaneous electroencephalography and fMRI recordings. MATERIALS AND METHODS: Fourteen children with West syndrome underwent simultaneous electroencephalography and fMRI at the onset of West syndrome. Statistically significant blood oxygen level-dependent responses related to hypsarrhythmia were analyzed using an event-related design of 4 hemodynamic response functions with peaks at 3, 5, 7, and 9 seconds after the onset of each event. Six of 14 children had focal epileptiform discharges after treatment and underwent simultaneous electroencephalography and fMRI from 12 to 25 months of age. RESULTS: At onset, positive blood oxygen level-dependent responses were seen in the brainstem (14/14 patients), thalami (13/14), basal ganglia (13/14), and hippocampi (13/14), in addition to multiple cerebral cortices. Group analysis using hemodynamic response functions with peaks at 3, 5, and 7 seconds showed positive blood oxygen level-dependent responses in the brainstem, thalamus, and hippocampus, while positive blood oxygen level-dependent responses in multiple cerebral cortices were seen using hemodynamic response functions with peaks at 5 and 7 seconds. In the remote period, 3 of 6 children had focal epileptiform discharge-related positive blood oxygen level-dependent responses in the thalamus, hippocampus, and brainstem. CONCLUSIONS: Positive blood oxygen level-dependent responses with hypsarrhythmia appeared in the brainstem, thalamus, and hippocampus on earlier hemodynamic response functions than the cerebral cortices, suggesting the propagation of epileptogenic activities from the deep brain structures to the neocortices. Activation of the hippocampus, thalamus, and brainstem was still seen in half of the patients with focal epileptiform discharges after adrenocorticotropic hormone therapy.

DOI： 10.3174/ajnr.A7646

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Language：English   Publisher：(一社)日本癌治療学会  

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Language：English   Publisher：(一社)日本癌治療学会  

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Language：Japanese   Publisher：(一社)日本神経学会  

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Language：Japanese   Publisher：(一社)日本神経学会  

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OBJECTIVE: After revascularization surgery in pediatric patients with moyamoya disease (MMD), resting and avoiding crying is important. However, this inaction is often difficult because of pain or anxiety. Dexmedetomidine (DEX), which has sedative and analgesic properties, may be useful in reducing those uncomfortable conditions; however, its common side effects include bradycardia and hypotension, which have a risk of decreasing the cerebral blood flow. The aim of this study was to investigate the efficacy and safety of using DEX for pediatric patients with MMD in the acute period after revascularization surgery. METHODS: This retrospective study included pediatric patients with MMD who underwent revascularization surgery. Based on whether DEX was used for light sedation during postoperative days (PODs) 0-1 after extubation, the patients were divided into DEX or control groups. For neurological outcomes, the incidence of symptomatic cerebral infarction and transient neurological events (TNEs) during PODs 0-1 and the entire hospitalization were investigated. In addition, the Richmond Agitation-Sedation Scale (RASS) was used to assess the effect of DEX, and bradycardia and hypotension were evaluated as side effects. RESULTS: A total of 84 surgical procedures were included in this study (27 in the DEX group and 57 in the control group). During PODs 0-1, symptomatic infarction was not observed in either group. The incidence of TNEs was almost the same in both groups: 2 (7.4%) of the 27 procedures in the DEX group and 4 (7.0%) of the 57 procedures in the control group (p > 0.99). Moreover, the incidences of symptomatic infarction and TNEs during the entire hospitalization did not differ significantly (symptomatic infarction, p > 0.99; TNEs, p = 0.20). Regarding the DEX effect, the median RASS scores during PODs 0-1 were -1.0 (drowsy) in the DEX group and +1.0 (restless) in the control group, showing a significant difference (p < 0.01). Regarding side effects, bradycardia was observed only in 3 (11.1%) of the 27 procedures in the DEX group (p = 0.03), and hypotension was not observed in any of the cases. CONCLUSIONS: In pediatric patients with MMD who are extubated after revascularization surgery, DEX produced appropriate light sedation and analgesia. The risk for symptomatic infarction is almost the same in cases in which DEX is used and those in which it is not; however, neurosurgeons should be cautious of bradycardia and TNEs as potential side effects.

DOI： 10.3171/2022.6.PEDS2241

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Language：English   Publisher：(一社)日本癌学会  

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Cervical angina, originating from cervical spine disorders, is a rare clinical syndrome presenting with chest pain mimicking angina pectoris. A rare case of cervical angina with cervical disc herniation requiring coronary artery stenting because of a true angina pectoris attack immediately after cervical spine surgery is reported. A 77-year-old woman presented with persistent pain around the neck, bilateral chest, left shoulder, and left back. She also complained of numbness and motor weakness in her left upper limb. Cervical spine imaging examinations showed instability at C4-5 and a calcified herniated disc with spinal cord compression at C4-6. She had a history of hypertension, diabetes mellitus not requiring insulin, and atrial fibrillation for which she was on anticoagulant therapy. The preoperative electrocardiogram and transthoracic echocardiography were within normal limits, and thus further cardiac study was considered unnecessary by the cardiologist. The anterior cervical discectomy and fusion were safely performed. However, she suddenly started to complain of left anterior chest pain with a cold sweat one hour after the surgery. An emergency electrocardiogram showed ischemic changes, and coronary angiography showed 99% stenosis at the right main coronary artery. A coronary stent was placed and good dilatation was achieved. Preoperative cervical angina symptoms such as numbness, motor weakness, and pain were improved immediately after surgery. The left chest pain also disappeared completely after coronary artery stent placement. Postoperative cervical imaging assessments showed good spinal decompression. She was discharged home without neurological deficits and no cardiac symptoms.

DOI： 10.7759/cureus.28313

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Language：Japanese   Publisher：一般社団法人 日本内分泌学会  

DOI： 10.1507/endocrine.98.s.hpt_25

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Language：Japanese   Publisher：一般社団法人 日本内分泌学会  

DOI： 10.1507/endocrine.98.s.hpt_72

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BACKGROUND: Although the risk of developing malignant lymphoma is higher in patients with rheumatoid arthritis (RA) than in the general population, primary central nervous system lymphoma (PCNSL) in patients with RA is extremely rare. In recent years, there has been concern that biological disease-modifying antirheumatic drugs (bDMARDs), widely administered to patients with RA, might increase the risk of cancer development. The authors report the first case of PCNSL in a patient with RA receiving the bDMARD tocilizumab. OBSERVATIONS: A 70-year-old man who was diagnosed with RA in 2010 was treated with low-dose methotrexate (MTX) from 2010 to 2015. Tocilizumab was commenced in 2012. In 2018, he developed gait disturbances, and gadolinium-enhanced magnetic resonance imaging showed multiple contrast-enhanced lesions in the basal ganglia and brain stem. Stereotactic brain biopsy led to the diagnosis of diffuse large B-cell lymphoma, and finally PCNSL was diagnosed. He was treated with five courses of MTX 3.5 g/m2, and his disease has been in remission for 34 months. LESSONS: Low-dose MTX and bDMARDs are associated with the concern of increased cancer risk in patients with RA. Because tocilizumab has been in use for a relatively short time, further accumulation of cases and careful follow-up are necessary.

DOI： 10.3171/CASE22256

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Perivascular spaces are fluid-filled spaces that surround the perforating vessels of the brain and are normal findings on brain imaging. These are usually asymptomatic and are considered a manifestation of aging. Perivascular spaces occasionally undergo significant enlargement and are referred to as tumefactive perivascular spaces, which are often indistinguishable from neoplastic lesions. Spontaneous regression of tumefactive perivascular spaces during follow-up is rare. We report the imaging findings and clinical course of a patient who showed spontaneous regression of tumefactive perivascular spaces in the anterior temporal lobe, together with a literature review and discussion regarding the characteristics and pathogenesis of spontaneous regression of tumefactive perivascular spaces. Most studies in the available literature report tumefactive perivascular spaces in the anterior temporal lobe; in our view, the characteristics of anterior temporal lobe tumefactive perivascular spaces may differ from those of tumefactive perivascular spaces that occur at other locations.

DOI： 10.18999/nagjms.84.3.678

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OBJECTIVE: We retrospectively compared the radiological and clinical outcomes of two different surgical techniques (lumbar spinous process splitting laminectomy [LSPSL] and unilateral laminotomy for bilateral decompression [ULBD]) to treat lumbar spinal canal stenosis (LCS). METHODS: We performed a retrospective comparative study of 141 consecutive patients with an average age of 70.8 ± 9.4 years who had undergone LSPSL or ULBD for LCS between April 2015 and April 2019. None of the patients had developed remote fractures of the spinous processes using either technique. These cases were divided into 2 groups: group L, 73 patients who had undergone LSPSL from April 2015 to April 2017; and group U, 68 patients who had undergone ULBD from May 2017 to April 2019. The clinical and radiological outcomes and surgical complications at the 1-year postoperative follow-up period were evaluated. RESULTS: We found no significant differences in the operative time between the 2 groups. However, group U had had significantly less blood loss than group L. The facet joints were significantly well preserved in group U. We examined the multilevel and spondylolisthesis cases separately and found that both surgical procedures were equally effective and that the visual analog scale scores for back or leg pain and Japanese Orthopaedic Association scores had significantly improved postoperatively in each group. Group U showed better outcomes in terms of LCS recurrence, with 3 patients in the group L requiring repeat surgery. CONCLUSIONS: We found both ULBD and LSPSL to be safe and effective techniques for LCS, even for patients with spondylolisthesis and multilevel disease. ULBD was superior in terms of recurrence prevention, preservation of the facet joints, and less blood loss.

DOI： 10.1016/j.wneu.2022.04.079

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OBJECTIVE: The parietooccipital fissure is an anatomical landmark that divides the temporal, occipital, and parietal lobes. More than 40% of gliomas are located in these three lobes, and the temporal lobe is the most common location. The parietooccipital fissure is located just posterior to the medial temporal lobe, but little is known about the clinical significance of this fissure in gliomas. The authors investigated the anatomical correlations between the parietooccipital fissure and posterior medial temporal gliomas to reveal the radiological features and unique invasion patterns of these gliomas. METHODS: The authors retrospectively reviewed records of all posterior medial temporal glioma patients treated at their institutions and examined the parietooccipital fissure. To clarify how the surrounding structures were invaded in each case, the authors categorized tumor invasion as being toward the parietal lobe, occipital lobe, isthmus of the cingulate gyrus, insula/basal ganglia, or splenium of the corpus callosum. DSI Studio was used to visualize the fiber tractography running through the posterior medial temporal lobe. RESULTS: Twenty-four patients with posterior medial temporal gliomas were identified. All patients presented with a parietooccipital fissure as an uninterrupted straight sulcus and as the posterior border of the tumor. Invasion direction was toward the parietal lobe in 13 patients, the occipital lobe in 4 patients, the isthmus of the cingulate gyrus in 19 patients, the insula/basal ganglia in 3 patients, and the splenium of the corpus callosum in 8 patients. Although the isthmus of the cingulate gyrus and the occipital lobe are located just posterior to the posterior medial temporal lobe, there was a significantly greater preponderance of invasion toward the isthmus of the cingulate gyrus than toward the occipital lobe (p = 0.00030, McNemar test). Based on Schramm's classification for the medial temporal tumors, 4 patients had type A and 20 patients had type D tumors. The parietooccipital fissure determined the posterior border of the tumors, resulting in a unique and identical radiological feature. Diffusion spectrum imaging (DSI) tractography indicated that the fibers running through the posterior medial temporal lobe toward the occipital lobe had to detour laterally around the bottom of the parietooccipital fissure. CONCLUSIONS: Posterior medial temporal gliomas present identical invasion patterns, resulting in unique radiological features that are strongly affected by the parietooccipital fissure. The parietooccipital fissure is a key anatomical landmark for understanding the complex infiltrating architecture of posterior medial temporal gliomas.

DOI： 10.3171/2021.7.JNS21990

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OBJECTIVES: Congenital afibrinogenemia is an autosomal recessive inherited disorder that can cause thrombotic as well as hemorrhagic events. We describe a case of repeated mild ischemic strokes due to a mural thrombus in the carotid artery and our medical treatment. CASE DESCRIPTION: A 49-year-old woman with congenital afibrinogenemia developed two minor ischemic strokes in two months. Clinical images revealed scattered fresh infarcts in the right middle cerebral artery region and mild cervical carotid artery stenosis. The risk for surgical treatment was considered to be extraordinarily high. The patient was treated with 100 mg/day of aspirin and 3 g fibrinogen infusion every two weeks. After the one-year course of medication, the mural thrombus gradually decreased, and there were no bleeding or ischemic stroke events. CONCLUSION: This case report highlights the successful treatment of an ischemic stroke in a patient with a congenital afibrinogenemia with an antiplatelet agent and fibrinogen replacement. There are no guidelines for managing ischemic stroke in patients with congenital afibrinogenemia, and further studies are needed.

DOI： 10.1016/j.jstrokecerebrovasdis.2022.106526

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DOI： 10.11477/mf.5002201799

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BACKGROUND: Eccrine spiradenocarcinoma (SC), also known as malignant eccrine spiradenoma, is a rare malignant cutaneous adnexal neoplasm arising from long-standing benign eccrine spiradenoma. Malignant skin tumors rarely show direct intracranial invasion. However, once the intracranial structure is infiltrated, curative excision with sufficient margins can become extremely difficult, particularly when the venous sinuses are involved. No effective adjuvant therapies have yet been established. Here, we report an extremely rare case of scalp eccrine SC with direct intracranial invasion, which does not appear to have been reported previously. CASE PRESENTATION: An 81-year-old woman presented with a large swelling on the parietal scalp 12 years after resection of spiradenoma from the same site. The tumor showed intracranial invasion with involvement of the superior sagittal sinus and repeated recurrences after four surgeries with preservation of the sinus. The histopathological diagnosis was eccrine SC. Adjuvant high-precision external beam radiotherapy (EBRT) proved effective after the third surgery, achieving remission of the residual tumor. The patient died 7 years after the first surgery for SC. CONCLUSIONS: Scalp SC with direct intracranial invasion is extremely rare. Radical resection with tumor-free margins is the mainstay of treatment, but the involvement of venous sinuses makes this unfeasible. High-precision EBRT in combination with maximal resection preserving the venous sinuses could be a treatment option for local tumor control.

DOI： 10.1186/s12883-022-02749-4

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Language：English   Publisher：LIPPINCOTT WILLIAMS & WILKINS  

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The purpose of this study was to examine the effects of combined revascularization for ischaemic-onset moyamoya disease (MMD) on cerebral haemodynamics by comparing cerebral blood flow (CBF) during the postoperative chronic phase with preoperative CBF. A retrospective cohort of 24 MMD patients (representing 31 surgeries) who received single photon emission computed tomography (SPECT) before and more than 6 months after surgery was investigated. The CBF value of each vascular territory was extracted from SPECT data, and the value relative to the ipsilateral cerebellar value (relative CBF, or RCBF) was calculated. The correlation between the revascularization effect and the proportional change in RCBF before and after surgery (calculated as post-RCBF/pre-RCBF ("post/pre-RCBF")) was analysed. Furthermore, the relationships between changes in neurological symptoms and post/pre-RCBF were investigated. Preoperative and postoperative mean RCBF values were 0.92 ± 0.15 and 0.96 ± 0.13 (p = 0.619) in the anterior cerebral artery territory, 0.99 ± 0.17 and 1.01 ± 0.17 (p = 0.598) in the middle cerebral artery territory and 1.15 ± 0.22 and 1.14 ± 0.19 (p = 0.062) in the posterior cerebral artery territory, respectively. No significant correlation was found between the revascularization score and post/pre-RCBF. The revascularization score and post/pre-RCBF were not significant predictors of worsening neurological symptoms postoperatively. No significant change in RCBF was observed in any vascular territory in the chronic phase after revascularization. Combined revascularization may assist in the redirection of blood flow from the internal to the external carotid system and contribute to CBF maintenance.

DOI： 10.1007/s10143-022-01774-8

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BACKGROUND: Dural suturing is one of the most robust reconstruction methods in transsphenoidal surgery; however, the technique is considered difficult, primarily due to the restricted range of needle movement. METHOD: We performed dural threading during transsphenoidal surgery by holding the needle with forceps as a hook and moving the needle in a distal to proximal direction. CONCLUSION: Our dural suturing technique is simple and quick. It can be used not only for dural closure but also for other procedures, such as controlling haemostasis from the intercavernous sinus and dural tenting.

DOI： 10.1007/s00701-022-05227-5

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BACKGROUND: We examined compliance differences among balloon remodeling microcatheters, which have not been established previously. METHODS: Straight and 120° angulated vascular models were created in a 3 mm diameter tube with 3 mm hole (vascular model A), a tube with a 4 mm hole (vascular model B), and a 4 mm diameter tube (vascular model C). We compared the pressure exerted when each balloon was herniated 1 or 2 mm between three compliant balloons (SHOURYU SR, TransForm C, and Scepter C) and four super-compliant balloons (HyperForm, SHOURYU HR, TransForm SC, and Scepter XC). RESULTS: In vascular model A, there was a significant difference in the pressure exerted by compliant balloons and super-compliant balloons in both the straight and angulated models. In the straight model (1 and 2 mm), the lowest pressure was exerted by HyperForm (super-compliant balloons group) and SHOURYU SR (compliant balloons group). The lowest pressure was exerted in the angulated model by HyperForm (super-compliant balloons group) and Scepter C (compliant balloons group). The Scepter balloon exerted higher pressure in the straight model than other balloon remodeling microcatheters but less in the angulated model. In vascular model B, the pressure decreased in all balloons compared with model A. In vascular model C, the pressure increased in all balloons compared with model A. CONCLUSIONS: Pressure differed across balloon remodeling microcatheters. In addition, vessel shape and diameter, and hole size, affected the results. Our findings can help select balloon remodeling microcatheters.

DOI： 10.1177/15910199211031765

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The study of genetic alterations and molecular biology in central nervous system (CNS) tumors has improved the accuracy of estimations of patient prognosis and tumor categorization. Therefore, the updated 2021 World Health Organization (WHO) classification includes various diagnostic genes, molecules, and pathways for diagnosis, as well as histological findings. These findings are expected both to have diagnostic applications and to facilitate new targeted therapies that target tumor-specific genetic changes and molecular biology. Intramedullary spinal cord tumors (IMSCTs) are rare CNS tumors that are difficult to treat because they occur in eloquent areas. Although the genetic underpinnings of IMSCTs remain unclear compared to their intracranial counterparts, the genetic characteristics of these tumors are gradually being revealed. Here, we describe the major changes in the new 2021 WHO classification and review the major types of IMSCTs, with an emphasis on their clinical features and genetic alterations.

DOI： 10.14245/ns.2244168.084

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Gliomas are a category of infiltrating glial neoplasms that are often located within or near the eloquent areas involved in motor, language, and neurocognitive functions. Surgical resection being the first-line treatment for gliomas, plays a crucial role in patient outcome. The role of the extent of resection (EOR) was evaluated, and we reported significant correlations between a higher EOR and better clinical prognosis of gliomas. However, recurrence is inevitable, even after aggressive tumor removal. Thus, efforts have been made to achieve extended tumor resection beyond contrast-enhanced mass lesions in magnetic resonance imaging (MRI)-defined areas, a process known as supratotal resection. Since it has been reported that tumor cells invade beyond regions visible as abnormal areas on MRI, imaging underestimates the true spatial extent of tumors. Furthermore, tumor cells have the potential to spread 10-20 mm away from the MRI-verified tumor boundary. The primary goal of supratotal resection is to maximize EOR and prolong the progression-free and overall survival of patients with gliomas. The available data, as well as our own work, clearly show that supratotal resection of gliomas is a feasible technique that has improved with the aid of awake functional mapping using intraoperative direct electrical stimulation. Awake brain mapping has enabled neurosurgeons achieve supratotal resection with favorable motor, language, and neurocognitive outcomes, ensuring a better quality of life in patients with gliomas.

DOI： 10.3389/fneur.2022.874826

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Balloon test occlusion (BTO) is an angiographic test to evaluate ischemic tolerance after permanent occlusion of an internal carotid artery (ICA). BTO can simulate ischemia caused by parent artery occlusion and can be used to select a suitable bypass surgery using specific criteria. On the other hand, a postoperative thrombus can form despite proper case selection, optimal radiological evaluation, and an appropriate surgical strategy. Postoperative ischemic complications related to perforating branches are clinically significant. This simulation study aimed to analyze postoperative flow characteristics and elucidate the cause of ischemic complications related to the perforating branch using computational fluid dynamics (CFD). An unexpected postoperative thrombus formation related to the perforating branch occurred after treating a giant aneurysm in the cavernous portion of the ICA in a patient. Three-dimensional digital subtraction angiography was used to acquire flow data and set up the CFD simulation. The flow simulations were performed at various bypass flow rates. The CFD analysis indicated flow stagnation in the ICA only when surgical treatment using a low-flow bypass graft was performed. Thrombus formation may lead to ischemic complications related to the perforating branch, such as the anterior choroidal artery. BTO did not reflect the influence of bypass blood flow. Therefore, recognizing that blood flow stagnation may occur and comprehensively deciding on the surgical strategy by CFD analysis can be helpful to prevent ischemic complications in patients with giant aneurysms.

DOI： 10.1038/s41598-022-11321-6

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Language：Japanese   Publisher：日本脳腫瘍病理学会  

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BACKGROUND: In patients with moyamoya disease (MMD) who receive superficial temporal artery (STA) to middle cerebral artery (MCA) bypass, hypoperfusion remote from the anastomosis site rarely occurs. Watershed shift due to direct bypass has been proposed as the mechanism; however, no report has confirmed this phenomenon using angiography. OBSERVATIONS: A 48-year-old man presented with transient weakness in his left arm. Angiography revealed severe bilateral stenosis of the MCAs and moyamoya vessels. The right anterior cerebral artery (ACA) had short stenosis at A2 but ample blood supply to the cortical area of the right ACA and MCA regions. The patient was diagnosed with MMD and received a single STA-MCA bypass. The next day, he had difficulty communicating, and a cerebral infarction away from the anastomosis site was identified. Perfusion examination revealed hyperperfusion around the direct bypass and hypoperfusion away from the anastomosis site. Angiography revealed bypass patency; however, the original anterograde flow of the right ACA decreased significantly at the stenosed point, indicating an improper watershed shift. LESSONS: STA-MCA bypass for patients with MMD can cause an improper watershed shift decreasing cerebral flow. Donor flow should be prepared based on each angiographic characteristic, and the risk of the improper watershed shift should be considered.

DOI： 10.3171/CASE22104

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Superficial temporal artery (STA) to superior cerebellar artery (SCA) bypass is usually performed via the subtemporal approach (StA), anterior transpetrosal approach (ApA), or combined petrosal approach (CpA), but no study has yet reported a quantitative comparison of the operative field size provided by each approach, and the optimal approach is unclear. The objective of this study is to establish evidence for selecting the approach by using cadaver heads to measure the three-dimensional distances that represent the operative field size for STA-SCA bypass. Ten sides of 10 cadaver heads were used to perform the four approaches: StA, ApA with and without zygomatic arch osteotomy (ApA-ZO- and ApA-ZO+), and CpA. For each approach, the major-axis length and the minor-axis length at the anastomosis site (La-A and Li-A), the major-axis length and the minor-axis length at the brain surface (La-B and Li-B), the depth from the brain surface to the anastomosis site (Dp), and the operating angles of the major axis and the minor axis (OAa and OAi) were measured. Shallower Dp and wider operating angle were obtained in the order CpA, ApA-ZO+, ApA-ZO-, and StA. In all parameters, ApA-ZO- extended the operative field more than StA. ApA-ZO+ extended La-B and OAa more than ApA-ZO-, whereas it did not contribute to Dp and OAi. CpA significantly decreased Dp, and widened OAa and OAi more than ApA-ZO+. ApA and CpA greatly expanded the operative field compared with StA. These results provide criteria for selecting the optimal approach for STA-SCA bypass in light of an individual surgeon's anastomosis skill level.

DOI： 10.1007/s10143-021-01686-z

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Language：Japanese   Publisher：Neurosurgical Review  

Trapped temporal horn is a rare type of noncommunicating focal hydrocephalus, and no standard treatment has been established yet for trapped temporal horn. Recent studies have shown the efficacy of endoscopic ventriculocisternostomy by opening the choroidal fissure; however, some surgical complications were reported during the procedure. Thus, we aimed to report a novel endoscopic ventriculocisternostomy and stenting technique for trapped temporal horn. In this technique, a 5.8-mm transparent acryl puncture needle with a 2.7-mm 0° rigid endoscope was used to open the choroidal fissure. It can fenestrate the choroidal fissure under real-time endoscopic observation of the critical neurovascular structures over the choroidal fissure. Moreover, the dull tip of the needle is less likely to injure the critical neurovascular structures, resulting in safer ventriculocisternostomy than the previously reported technique. Then, a stent is placed along the tract to prevent future obstruction of the stoma. Six trapped temporal horns in four patients were treated using the technique. All the patients showed improved symptoms with no surgical complications. None of the patients showed recurrence of trapped temporal horn during the mean follow-up period of 39.3 months. The combination of endoscopic ventriculocisternostomy and stenting with a transparent acryl puncture needle is a safe and effective treatment option for trapped temporal horn.

DOI： 10.1007/s10143-021-01634-x

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Cerebral revascularization for moyamoya disease (MMD) is an effective treatment for improving cerebral ischaemia and preventing rebleeding. Although direct bypass surgery is commonly performed on older children and adults, it is challenging in very young children due to the high difficulty level of the procedure. The subjects were MMD patients under 3 years of age on whom surgery was performed by a single surgeon (Y.A.). Preoperative clinical findings, information related to direct bypass surgery, bypass patency, and the incidence of postoperative stroke were investigated. Combined revascularization, including direct bypass surgery, was performed on 3 MMD patients (3 sides) under 3 years of age. The average diameter of the grafts used in direct bypass was 0.8 mm. The average recipient diameter was 0.8 ± 0.17 (range 0.6-1) mm. In all cases, the anastomotic procedure was completed using 11-0 monofilament nylon thread, and patency was confirmed. Direct bypass for MMD patients under 3 years old is technically challenging. However, despite the anatomical differences between very young children and elderly individuals, direct bypass surgery could certainly be completed. In addition, a rapid recovery from cerebral blood flow insufficiency could yield a promising neurological outcome.

DOI： 10.1007/s10143-021-01685-0

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OBJECTIVE: Cerebral revascularization is necessary for pediatric patients younger than 5 years with moyamoya disease (MMD). However, they have a high risk of developing cerebral infarction early after surgery. This study aimed to analyze the risk factors for developing cerebral infarction among these patients. METHODS: The charts of 21 consecutive patients with MMD (39 surgeries) younger than 5 years who had undergone revascularization at our hospital were retrospectively analyzed. Because cerebral infarction occurring within 1 month after surgery was the primary end point, other clinical information was evaluated, including each surgical procedure. Multivariate analysis of the risk factors for postoperative cerebral infarction was performed. RESULTS: Cerebral infarction occurred after 7 of 39 surgeries (17.9%). Of the 39 surgeries, 23 (59%) included direct and indirect combined revascularization. The incidence of cerebral infarction did not differ significantly between the combined (21.7%) and indirect (12.5%) groups (P = 0.46). Logistic regression showed no association between the revascularization procedure and the occurrence of cerebral infarction after surgery (P = 0.3). However, younger age at surgery was correlated with a higher risk of developing cerebral infarction in the early postoperative period (P = 0.05). CONCLUSIONS: No differences were found in the risk of developing cerebral infarction early after surgery as a result of surgical procedures. However, younger patients had higher postoperative risk. Further multicenter research should examine this issue for young pediatric patients with moyamoya at high risk of developing cerebral infarction.

DOI： 10.1016/j.wneu.2021.12.115

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Language：English   Publishing type：Research paper (scientific journal)   Publisher：The Japan Neurosurgical Society  

Extended endonasal transsphenoidal surgery (eTSS) offers a wide surgical field for various parasellar lesions; however, intraoperative high-flow cerebrospinal fluid (CSF) leakage is inevitable. Therefore, secure sellar reconstruction methods are essential to prevent postoperative CSF leakage. Although collagen matrix has been applied for dural reconstruction in neurosurgery, its suitability for application in extended eTSS remains unclear. Eighteen patients underwent modified shoelace dural closure using collagen matrix after lesionectomy via extended eTSS. In this technique, a collagen matrix, which was placed subdurally (inlay graft), was continuously sutured with both open dural edges like a shoelace. Then, another collagen matrix was placed epidurally (onlay graft), and rigid reconstruction was performed using the septal bone and a resorbable fixation mesh. Postoperative CSF leakage did not occur in 17 patients but did occur in 1 patient with tuberculum sellae meningioma. In this case, the CSF leakage point was detected just around the area between the coagulated dura and the adjacent collagen matrix. The collagen matrix harvested from this area was pathologically examined; neovascularization and fibroblastic infiltration into the collagen matrix were not detected. On the other hand, neovascularization and fibroblast infiltration into the collagen matrix were apparent on the surface of the collagen matrix harvested from the non-CSF leakage area. Our novel dural closure technique using collagen matrix could be an effective option for sellar reconstruction in extended eTSS; however, it should be applied in patients in whom normal dural edges are preserved.

DOI： 10.2176/jns-nmc.2021-0355

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Language：Japanese   Publisher：(一社)日本小児神経外科学会  

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Language：Japanese   Publisher：(一社)日本内分泌学会  

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Language：Japanese   Publisher：(一社)日本内分泌学会  

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Language：Japanese   Publisher：(一社)日本内分泌学会  

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Publisher：三輪書店  

DOI： 10.11477/mf.5002201765

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BACKGROUND: Previous studies showed that although the risk of thyroid dysfunction [thyroid immune-related adverse events (irAEs)] induced by anti-programmed cell death-1 antibodies (PD-1-Ab) was as low as 2% to 7% in patients negative for anti-thyroid antibodies (ATAs) at baseline, it was much higher (30%-50%) in patients positive for ATAs. However, whether a similar increase occurs with combination therapy using PD-1-Ab plus anti-cytotoxic T-lymphocyte antigen-4 antibody (CTLA-4-Ab) is unknown. METHODS: A total of 451 patients with malignancies treated with PD-1-Ab, CTLA-4-Ab, or a combination of PD-1-Ab and CTLA-4-Ab (PD-1/CTLA-4-Abs) were evaluated for ATAs at baseline and for thyroid function every 6 weeks for 24 weeks after treatment initiation and then observed until the last clinical visit. RESULTS: Of the 451 patients, 51 developed thyroid irAEs after immunotherapy [41 of 416 (9.9%) treated with PD-1-Ab, 0 of 8 (0%) treated with CTLA-4-Ab, and 10 of 27 (37.0%) treated with PD-1/CTLA-4-Abs]. The cumulative incidence of thyroid irAEs was significantly higher in patients who were positive vs negative for ATAs at baseline after both PD-1-Ab [28/87 (32.2%) vs 13/329 (4.0%), P < 0.001] and PD-1/CTLA-4-Abs [6/10 (60.0%) vs 4/17 (23.5%), P < 0.05] treatments. The risk of thyroid irAEs induced by PD-1/CTLA-4Abs, which was significantly higher than that induced by PD-1-Ab, in patients negative for ATAs at baseline was not statistically different from that induced by PD-1-Ab in patients positive for ATAs at baseline. CONCLUSIONS: This study showed that the incidence of thyroid irAEs was high and not negligible after PD-1/CTLA-4-Abs treatment even in patients negative for ATAs at baseline.

DOI： 10.1210/clinem/dgab829

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BACKGROUND: Remote cerebral infarction after combined revascularization of the middle cerebral artery (MCA) territory is rare in patients with moyamoya disease (MMD) with a fetal-type posterior communicating artery (PCoA). OBSERVATIONS: A 57-year-old woman developed numbness in her right upper limb and transient motor weakness and was diagnosed with MMD. She also had a headache attack and a scintillating scotoma in the right visual field. Preoperative magnetic resonance angiography (MRA) showed stenosis of the left posterior cerebral artery (PCA). Combined revascularization was performed for the left MCA territory. No new neurological deficits were observed for 2 days after the operation, but right hemianopia, alexia, and agraphia appeared on postoperative day (POD) 4. Magnetic resonance imaging showed a new left occipitoparietal lobe infarction, and MRA showed occlusion of the distal left PCA. After that point, the alexia and agraphia gradually improved, but right hemianopia remained at the time of discharge on POD 18. LESSONS: Cerebral ischemia in the PCA territory may occur after combined revascularization of the MCA territory in patients with fetal-type PCoA. For these cases, a double-barrel bypass or indirect revascularization to induce a slow conversion could be considered on its own as a treatment option.

DOI： 10.3171/CASE21704

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Language：English   Publishing type：Research paper (scientific journal)   Publisher：Journal of Neurosurgery Publishing Group (JNSPG)  

BACKGROUND

Adenoid cystic carcinoma (ACC) of the external auditory canal (EAC) is a rare tumor that accounts for approximately 5% of all EAC tumors. ACC is generally known as a slow-growing tumor, but patients often experience recurrence or distant metastasis in the long clinical course. While the major pattern of recurrence is pulmonary metastasis, brain metastasis of ACC of the EAC is rare.

OBSERVATIONS

The authors describe the case of a 72-year-old male who was diagnosed with ACC of the EAC. Approximately 7 years later, brain magnetic resonance imaging revealed an intra-axial homogenously enhancing mass lesion that had no direct connection with the skull base in the left frontal lobe. The patient underwent tumor resection and histopathological examination revealed a mixture of cribriform and tubular patterns. The image and pathological characteristics of the tumor were similar to those of primary ACC or ACC from other sites of origin.

LESSONS

While patients with ACC of the EAC often experience recurrence or distant metastasis in the long clinical course, they survive for a relatively long period of time, even though an optimal treatment has not been established. The authors therefore recommend surgical resection for brain metastasis of ACC of the EAC to improve neurological symptoms.

DOI： 10.3171/CASE21673

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Other Link： https://thejns.org/downloadpdf/journals/j-neurosurg-case-lessons/3/11/article-CASE21673.xml

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OBJECTIVE: Magnetic resonance-guided focused ultrasound (MRgFUS) thalamotomy ameliorates symptoms in patients with essential tremor (ET). How this treatment affects canonical brain networks has not been elucidated. The purpose of this study was to clarify changes of brain networks after MRgFUS thalamotomy in ET patients by analyzing resting-state networks (RSNs). METHODS: Fifteen patients with ET were included in this study. Left MRgFUS thalamotomy was performed in all cases, and MR images, including resting-state functional MRI (rsfMRI), were taken before and after surgery. MR images of 15 age- and sex-matched healthy controls (HCs) were also used for analysis. Using rsfMRI data, canonical RSNs were extracted by performing dual regression analysis, and the functional connectivity (FC) within respective networks was compared among pre-MRgFUS patients, post-MRgFUS patients, and HCs. The severity of tremor was evaluated using the Clinical Rating Scale for Tremor (CRST) score pre- and postoperatively, and its correlation with RSNs was examined. RESULTS: Preoperatively, ET patients showed a significant decrease in FC in the sensorimotor network (SMN), primary visual network (VN), and visuospatial network (VSN) compared with HCs. The decrease in FC in the SMN correlated with the severity of tremor. After MRgFUS thalamotomy, ET patients still exhibited a significant decrease in FC in a small area of the SMN, but they exhibited an increase in the cerebellar network (CN). In comparison between pre- and post-MRgFUS patients, the FC in the SMN and the VSN significantly increased after treatment. Quantitative evaluation of the FCs in these three groups showed that the SMN and VSN increased postoperatively and demonstrated a trend toward those of HCs. CONCLUSIONS: The SMN and CN, which are considered to be associated with the cerebello-thalamo-cortical loop, exhibited increased connectivity after MRgFUS thalamotomy. In addition, the FC of the visual network, which declined in ET patients compared with HCs, tended to normalize postoperatively. This could be related to the hypothesis that visual feedback is involved in tremor severity in ET patients. Overall, the analysis of the RSNs by rsfMRI reflected the pathophysiology with the intervention of MRgFUS thalamotomy in ET patients and demonstrated a possibility of a biomarker for successful treatment.

DOI： 10.3171/2022.5.JNS22411

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BACKGROUND: Transient neurological deficits (TNDs) develop after cerebral revascularization in patients with moyamoya disease (MMD). The authors report a rare pediatric MMD case with extensive decreased cerebral blood flow (CBF) and prolonged TNDs after combined revascularization. OBSERVATIONS: A 9-year-old boy presented with transient left upper limb weakness, and MMD was diagnosed. A right-sided combined surgery was performed. Two years after the surgery, frequent but transient facial (right-sided) and upper limb weakness appeared. The left internal carotid artery terminal stenosis had progressed. Therefore, a left combined revascularization was performed. The patient's motor aphasia and right upper limb weakness persisted for approximately 10 days after surgery. Magnetic resonance angiography showed that the direct bypass was patent, but extensive decreases in left CBF were observed using single photon emission tomography. With adequate fluid therapy and blood pressure control, the neurological symptoms eventually disappeared, and CBF improved. LESSONS: The environment of cerebral hemodynamics is heterogeneous after cerebral revascularization for MMD, and the exact mechanism of CBF decreases was not identified. TNDs are significantly associated with the onset of stroke during the early postoperative period. Therefore, appropriate treatment is desired after determining complex cerebral hemodynamics using CBF studies.

DOI： 10.3171/CASE21628

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Diffuse midline glioma(DMG), H3 K27M-mutant is an infiltrative midline high-grade glioma with predominantly astrocytic differentiation and a K27M mutation in either H3F3A or HIST1H3B/C. It is commonly located in the brain stem, thalamus, and spinal cord. DMG is predominant in children but can occur in adults. Brain stem disease, known as diffuse intrinsic pontine glioma(DIPG), is the representative: -80% harbor the H3 K27M mutation. Generally, the prognosis of DMG is poor: the 2-year survival rate is < 10%, despite intensive research and therapies. Consequently, radiation is the only treatment and there is no effective chemotherapeutic regimen. The recent findings concerning the genetic profiles of DMG shed light on precision medicine. Until today, approximately 250 clinical trials with molecular targeted therapy as a strategy have been conducted for different biological pathways in DMG. Unfortunately, none of them has shown significant efficacy for DMG. One of the problems in these clinical trials is insufficient knowledge of whether the used molecular targeted agents penetrate the blood-brain barrier. Continuous efforts to develop effective precision medicine against DMG should pave the way for overcoming DMG in the future.

DOI： 10.11477/mf.1436204529

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Language：English   Publishing type：Research paper (scientific journal)   Publisher：The Japan Neurosurgical Society  

Resting-state functional MRI (rs-fMRI) has been utilized to visualize large-scale brain networks. We evaluated the usefulness of multitier network analysis using rs-fMRI in patients with focal epilepsy. Structural and rs-fMRI data were retrospectively evaluated in 20 cases with medically refractory focal epilepsy, who subsequently underwent surgery. First, structural changes were examined using voxel-based morphometry analysis. Second, alterations in large-scale networks were evaluated using dual-regression analysis. Third, changes in cortical hubs were analyzed and the relationship between aberrant hubs and the epileptogenic zone (EZ) was evaluated. Finally, the relationship between the hubs and the default mode network (DMN) was examined using spectral dynamic causal modeling (spDCM). Dual-regression analysis revealed significant decrease in functional connectivity in several networks including DMN in patients, although no structural difference was seen between groups. Aberrant cortical hubs were observed in and around the EZ (EZ hubs) in 85% of the patients, and a strong degree of EZ hubs correlated to good seizure outcomes postoperatively. In spDCM analysis, facilitation was often seen from the EZ hub to the contralateral side, while inhibition was seen from the EZ hub to nodes of the DMN. Some cognition-related networks were impaired in patients with focal epilepsy. The EZ hub appeared in the vicinity of EZ facilitating connections to distant regions in the early phase, which may eventually generate secondary focus, while inhibiting connections to the DMN, which may cause cognitive deterioration. Our results demonstrate pathological network alterations in epilepsy and suggest that earlier surgical intervention may be more effective.

DOI： 10.2176/nmc.oa.2021-0173

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Since the World Health Organization 2016 classification (2016 WHO), genetic status has been incorporated into the diagnosis of Grade 2/3 gliomas (lower-grade gliomas). Therefore, immunohistochemistry (IHC) of IDH1-R132H, ATRX, and p53 have been used in place of genetic status. We report the associations between histological findings, IHC, and genetic status. We performed IHC of IDH1-R132H, ATRX, and p53 in 76 lower-grade gliomas and discussed its validity based on the 2016 WHO and the upcoming 2021 WHO classification. The sensitivity and specificity of anti-ATRX, p53, and IDH1-R132H IHC were 40.9%/98.1%, 78.6%/85.4%, and 90.5%/84.6%, respectively. Among 21 IDH1-mutant gliomas without 1p/19q codeletion, two gliomas (9.5%) mimicked the so-called classic for oligodendroglioma (CFO) in their morphology. Of the 42 gliomas with 1p/19q codeletion, four cases were difficult to diagnose as oligodendroglioma through morphological examination. Moreover, there were three confusing cases with ATRX mutations but with retained ATRX-IHC positivity. The lessons learned from this study are as follows: (1) ATRX-IHC and p53-IHC should be supplementary to morphological diagnosis, (2) rare IDH mutations other than IDH1 R132H should be considered, and (3) there is no complete alternative test to detect molecular features of glioblastoma under the 2021 WHO classification.

DOI： 10.1007/s10014-021-00418-x

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Language：Japanese   Publisher：The Japan Stroke Society  

<p>Here, we report a case of acute MCA occlusion in a patient who underwent thoracic endovascular aortic repair (TEVAR) for a thoracic aortic aneurysm 2 years ago. An 83-year-old man presented with sudden right hemiparesis and aphasia. CT with contrast revealed occlusion of the left MCA, and MRI revealed mild hyperintense signals in the left MCA area. The aortic arch stent graft partially overlapped the origin of the left common carotid artery on the contrast CT. Therefore, we decided not to perform mechanical thrombectomy via the transaortic approach. Instead, the patient underwent a surgical embolectomy with frontotemporal craniotomy. His symptoms improved remarkably, and he was discharged with only mild aphasia, dysarthria, and disorientation. In a case where mechanical thrombectomy via the transaortic approach is expected to be difficult, surgical embolectomy can be an alternative choice.</p>

DOI： 10.3995/jstroke.11028

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Authorship：Last author   Language：English   Publisher：The Japanese Society for Neuroendovascular Therapy  

OBJECTIVE: Owing to the limited time since the introduction of the PulseRider (PR), inconsequential or rare complications that clinicians should be aware of remain unreported yet. Here, we report a rare complication of incomplete detachment. CASE PRESENTATION: A 50-year-old male underwent PR-assisted coil embolization for a basilar tip aneurysm. Coiling was completed, and the detachment procedure was performed using a detachment machine; the success signal was observed. The delivery microcatheter was subsequently advanced back up to the proximal markers, and no reapproximation of the proximal markers, which indicates successful detachment, was observed. However, only one of the proximal markers returned to the microcatheter, and incomplete detachment of only one leg was detected. Ultimately, electrical detachment was not possible, and physical separation by tension was achieved. CONCLUSION: Our case report presents a rare case of a detachment problem in the PR. The PR could not be detached, although the signal revealed successful detachment. Therefore, careful withdrawal of the delivery wire by checking not only the proximal markers but also the behavior of the entire PR and coil complex is important.

DOI： 10.5797/jnet.cr.2021-0095

Web of Science

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Authorship：Last author   Language：English   Publisher：The Japanese Society for Neuroendovascular Therapy  

OBJECTIVE: Superior petrosal sinus dural arteriovenous fistula (SPS-DAVF) is a rare subtype of intracranial DAVF that sometimes leads to hemorrhagic symptoms following deep venous drainage. Here we report the case of SPS-DAVF with retrograde venous reflux to the cerebellar vein. Preoperative contrast-enhanced MRI was a decisive factor in a safe and effective treatment. CASE PRESENTATION: A 37-year-old woman was referred to our hospital with abnormal MRI findings, which was performed when she had a mild headache during her check-up. DSA revealed left-sided SPS-DAVF, which was diagnosed as Cognard type IIb. Both CTA and DSA could not detect the whole SPS but only the shunt pouch. Using contrast-enhanced MRI, we were able to visualize the presence of the SPS and its continuity within the shunt pouch. 3D-T1 turbo spin echo (SPACE) showed a low-intensity area in the SPS, which was not seen in the 3D-T1 fast field echo (FFE). During the procedure, there was a point where it was difficult to advance the microcatheter, which coincided with the low-intensity area. We achieved effective transvenous embolization from the occluded venous access by devising a surgical technique. CONCLUSION: In addition to the contrast-enhanced 3D-T1 FFE, 3D-T1 SPACE might provide beneficial information for endovascular therapy in the evaluation of venous sinuses, which could not be detected by standard examinations.

DOI： 10.5797/jnet.cr.2021-0029

Web of Science

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Language：Japanese   Publisher：The Japanese Society of Spinal Surgery  

<p>  The patient was a 46-year-old man who presented with fatigue for 6 months. His activities of daily living (ADL) gradually worsened and he was transported to our hospital’s emergency room because of difficulty in walking due to muscle weakness in the extremities. His weight had decreased by 20 kg in 6 months, and he was almost bedridden. Upon arrival, he presented with emaciation, hypotension, and muscle weakness predominantly in the proximal portions. However, he showed no clumsiness in the hands, pain, sensory disorders, or bladder-rectal disturbances. Only Hoffman’s reflex was positive. Deep tendon reflexes were almost normal in his upper limbs but flaccid in his lower limbs. The Japan Orthopaedic Association (JOA) score was 14/17. Images of the cervical spine showed a developmental narrow canal, cervical spondylotic changes at C4-7 levels, and cervical spinal cord compression with myelomalacia at C4-6 levels. These findings suggested cervical spondylotic myelopathy (CSM). However, laboratory data showed mild hyponatremia (sodium 135 mmol/L) and hypothyroidism (free thyroxine [T4] 1.00 ng/ml, thyroid-stimulating hormone [TSH] 6.62 µIU/ml). Based on radiology, endocrinology, and neurology consultations, the patient was diagnosed with an isolated adrenocorticotropic hormone (ACTH) deficiency. Hydrocortisone (20 mg/day) administration dramatically improved his symptoms and ADL. The JOA score improved to 17/17 over a few months, and the patient returned to his daily life. He has been followed-up without surgery until now.</p><p>  Several differential diagnoses other than CSM involve muscle weakness of the extremities. Even if there is a clear cervical spine lesion on imaging, it is important to consider whether it is a lesion actually responsible for the symptoms. Cooperation with other departments is also necessary. In particular, if a patient has endocrine diseases, such as in this case, it can cause fatal complications during surgery. Therefore, we should pay attention to these patients and initiate prophylactic measures if the patient needs surgery.</p>

DOI： 10.2531/spinalsurg.36.88

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Language：English  

DOI： 10.25259/SNI_453_2022

PubMed