Language：English   Publishing type：Research paper (scientific journal)  

INTRODUCTION: Recent studies have reported that essential tremor (ET) presents with not only motor symptoms but also cognitive dysfunction. However, detailed pathological mechanisms remain unclear. Here, we evaluate the characteristics of cognitive changes in older patients. METHODS: Eighty-five patients aged 65 years or older with ET but without dementia were evaluated for cognitive function using the Addenbrooke Cognitive Examination Revised (ACE-R). The patients were compared with healthy controls (HCs), and the characteristics of cognitive dysfunction were examined. Age at onset and correlations with tremor severity were also investigated. Moreover, we performed resting-state network (RSNs) analysis in a subset of these patients, and the functional connectivity (FC) within the networks was compared with age-matched controls. RESULTS: Compared to HCs, older patients with ET showed a clear reduction in the total (p = 0.001), attention (p = 0.005), verbal fluency (p = 0.001), and memory (p = 0.001) ACE-R scores. Older-onset patients showed significant cognitive dysfunction compared with younger-onset patients. Verbal fluency correlated with tremor severity in the multiple regression analysis (p < 0.001). RSNs showed an increase in FC in the frontal lobes within the language network in patients with ET compared to HCs (p < 0.05, FWE-corrected). CONCLUSION: Older patients with ET showed obvious cognitive dysfunction compared to HCs, indicating that cognitive dysfunction varies by age of onset and correlates with tremor severity. The results of the RSNs analysis suggest that the pathological mechanism of cognitive dysfunction in ET patients involves network changes similar to those in the early stages of Alzheimer's disease.

DOI： 10.1016/j.parkreldis.2024.107228

Web of Science

PubMed

researchmap

Language：English  

DOI： 10.1016/j.jstrokecerebrovasdis.2024.108202

Web of Science

PubMed

Language：English   Publishing type：Research paper (scientific journal)  

OBJECTIVE: This study examined the clinical significance of residual hyperintense area on T2-weighted magnetic resonance imaging (T2R) without gadolinium-enhanced lesions at the end of initial treatment (debulking surgery, concomitant radiotherapy, and temozolomide) in patients with glioblastoma. METHODS: Among 150 glioblastoma cases, 77 cases without enhanced lesions at the end of initial treatment and without factors modifying the distribution of residual hyperintense area or pattern of recurrence were included. We retrospectively reviewed the relationship of residual hyperintense area after initial treatment with progression-free survival (PFS), overall survival (OS), and pattern of recurrence. RESULTS: In these 77 cases, the median PFS and OS were 12.4 and 27.4 months, respectively. At the end of initial treatment, 55 (71.4%) cases had residual hyperintense area (T2 residual group, T2R), whereas 22 (28.6%) showed no hyperintense area (T2 disappeared group, disappeared hyperintense area on T2-weighted magnetic resonance imaging [T2D]). Based on univariate and multivariate analyses, the residual hyperintense area after initial treatment was not a prognostic factor for PFS or OS. Distant recurrences occurred more frequently in the T2D group than in the T2R group 50.0% versus 9.5%. In the T2R group, the recurrence site coincided with the residual hyperintense area in 36 (85.7%) of 42 recurrences. CONCLUSIONS: The T2R at the end of initial treatment can predict local recurrence. However, the distant recurrence occurred frequently in T2D group. Thus, attention should be paid to local recurrences in T2R group and distant recurrences in T2D group.

DOI： 10.1016/j.wneu.2024.10.012

Web of Science

PubMed

researchmap

Language：English   Publishing type：Research paper (scientific journal)  

Surgical interventions for arachnoid cysts and glioependymal cysts primarily focus on cyst decompression and establishing communication with cerebrospinal fluid (CSF) spaces. However, a standardized surgical strategy for symptomatic convexity cysts lacking surrounding CSF space is still lacking due to the limited number of cases. This systematic review aims to evaluate surgical interventions for symptomatic convexity cysts and proposes a safe and effective treatment approach we have developed. A systematic review of the literature was conducted following PRISMA guidelines. Surgical techniques were categorized into cystoperitoneal (CP) shunt, cyst excision, cyst fenestration, cyst ventriculostomy, and several uniquely modified methods. Using a method different from these previous reports, we performed endoscopic cystoventricular stenting via burr hole in three cases of convexity cysts. While traditional methods have proven effective, CP shunt may lead to malfunction, infection, and subdural hematoma; wall excision is invasive and increases morbidity risks. Fenestration and cystoventriculostomy have also reported cases of recurrence. Other methods also pose issues, including off-label use of instruments and inadequate consideration for removal. The method we performed for placing the stent catheter with multiple side holes demonstrated long-term effectiveness. Furthermore, the surgery was conducted with the patient fully awake, and the use of electrical stimulation mapping enabled preservation of higher neurocognitive functions, including language, working memory, and spatial cognition, at the puncture site. This novel approach offers advantages such as minimally invasive procedure, preservation of safe brain function, and long-term patency. Further studies are warranted to validate these findings and improve surgical strategies for convexity cysts.

DOI： 10.1016/j.wneu.2024.10.031

Web of Science

PubMed

researchmap

Language：English  

DOI： 10.1007/s10147-024-02650-9

Web of Science

PubMed

Publisher：三輪書店  

DOI： 10.11477/mf.5002202449

CiNii Research

Language：Japanese   Publisher：The Japan Society of Neurotraumatology  

<p>Although various types of vascular disorders are associated with head trauma, a rare case is a traumatic middle meningeal artery pseudoaneurysm (tMMAP). We report a case of a 70–year–old man with tMMAP presenting with an intracerebral hematoma (ICH). He was rushed to the hospital in a poor state of consciousness after a traumatic fall. A computer tomography (CT) scan taken on ad­mis­sion revealed a traumatic left temporal lobe ICH, subarachnoid hemorrhage, and bilateral acute subdural hematoma. After conservative treatment, his consciousness improved, except for disorientation on day 3 of hospitalization, and a CT scan revealed resolution of ICH. However, on day 10 of hospitalization, he suddenly fell into a coma, and a CT scan revealed that the hematoma in the left temporal lobe had grown and caused a midline shift. We conducted an emergency craniotomy and hematoma removal, during which we encountered an intractable arterial bleeding at the left middle skull base. We could barely stop the bleeding using aneurysmal clips. Suspecting the possibility of a vascular abnormality, we per­formed a postoperative cerebral angiography that revealed a pseudoaneurysm in the left middle meningeal artery. We determined that this pseudoaneurysm was the source of the bleeding and performed an endovascular parent vessel occlusion. After the endovascular surgery, there was no recurrence of hemorrhage. The patient was transferred to a rehabilitation facility because of residual cognitive dysfunction. tMMAP is a rare disorder, and its rupture generally causes an epidural hematoma; however, in some cases, it can cause an ICH. Although it is difficult to predict the rupture in advance, tMMAP must be recognized as a delayed com­plication of traumatic vascular disorders as it can lead to severe conditions.</p>

DOI： 10.32187/neurotraumatology.47.2_94

CiNii Research

Language：English   Publishing type：Research paper (scientific journal)  

BACKGROUND: Early seizures after craniotomy are significant perioperative complications that can adversely impact patient outcomes. Despite current guidelines advising against the routine use of antiseizure drugs for seizure after craniotomy prevention due to limited efficacy data, many clinicians continue prescribing them. This discrepancy highlights the need for robust evidence to guide clinical practice. This multi-center, randomized clinical trial was designed to investigate the efficacy of perampanel in preventing early seizures after craniotomy. METHOD: This multi-center, open-label, randomized clinical trial will be conducted across five hospitals in Nagoya, Japan, from February 2024 to December 2026. A total of 142 seizure-naive patients with supratentorial brain tumors will be recruited and randomized (1:1) into the treatment and control groups. The treatment group will receive 2 mg of perampanel starting 2 days preoperatively and continuing for 28 days postoperatively, while the control group will receive no antiseizure drugs. The primary outcome is the incidence of seizures within 28 days after craniotomy. Secondary outcomes are length of hospital and intensive care unit stays and postoperative complications. DISCUSSION: This study addresses the critical need for evidence-based recommendations regarding antiseizure drug use for preventing early seizures after craniotomy. As the first multi-center, randomized trial evaluating perampanel's efficacy in this setting, the findings may significantly influence clinical guidelines and perioperative practices. TRIAL REGISTRATION: This trial was registered with the Japan Registry of Clinical Trials (approval number: jRCTs041230117) on December 18, 2023, a member of the Primary Registry Network of the World Health Organization's International Clinical Trials Registry Platform.

DOI： 10.1186/s13063-024-08693-7

Open Access

Web of Science

Scopus

PubMed

researchmap

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,b a 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

Web of Science

Scopus

PubMed

Language：English  

DOI： 10.7759/cureus.76653

PubMed

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

DOI： 10.7759/cureus.74971

PubMed

researchmap

Language：English  

DOI： 10.1007/s00276-024-03522-7

Web of Science

PubMed

Language：English   Publishing type：Research paper (scientific journal)  

BACKGROUND: Abrupt discontinuation of overused medications is standard treatment for medication overuse headache (MOH), but discontinuation is difficult to maintain. The aim was to evaluate the real-world clinical results of anti-calcitonin gene-related peptide monoclonal antibody (CGRP-mAb) treatment for migraine with MOH without abrupt drug discontinuation and no hospitalization. METHODS: Data were collected before starting CGRP-mAb injections (baseline) and 1 month after each injection. The following items were compared between baseline and after the first, second, and third CGRP-mAb injections, monthly headache days (MHD), monthly migraine days (MMD), monthly acute medication use (AMU) days, monthly total amount of AMU tablets, headache impact test-6 (HIT-6), and the migraine-specific quality of life questionnaire (MSQ). Achieving reduction rates ≥50 % in the frequency of each headache and migraine was defined as a good response. Achieving reduction rates of both AMU days and tablets ≥50 % was defined as effective in reducing AMU. RESULTS: This study included 33 patients with migraine with MOH. After the third CGRP-mAb injection, MHD and MMD were significantly decreased from median 30.0 to 9.5 days, and 10.0 to 1.5 days, respectively. In addition, monthly AMU days and tablets were significantly decreased from median 28.0 to 8.0 days, and 30.0 to 9.5 tablets, respectively. After the third CGRP-mAb injection, the good MHD and MMD responder rates were 75.0 % and 85.7 %, respectively. The rate of reducing AMU was 78.6 %. HIT-6 and MSQ scores decreased significantly from baseline to after each CGRP-mAb injection. CONCLUSIONS: When CGRP-mAb was administered to migraine with MOH, frequency of headache symptoms and AMU were reduced without abrupt drug discontinuation and no hospitalization.

DOI： 10.1016/j.heliyon.2024.e40190

Open Access

Scopus

PubMed

researchmap

Language：English   Publishing type：Research paper (scientific journal)   Publisher：Japanese Society for Magnetic Resonance in Medicine  

<p>Purpose: To re-evaluate images recovered from JCOG0911, a randomized phase 2 trial for newly diagnosed glioblastoma (nGBM) conducted by the Japan Clinical Oncology Group (JCOG) Brain Tumor Study Group.</p><p>Methods: The correlation between tumor volumes and survival was evaluated, followed by progression-free survival (PFS) analysis by independent central review based on Response Assessment in Neuro-Oncology (RANO) criteria using MRI recovered from 118 nGBM patients enrolled in the JCOG0911 trial. A radiomic analysis was also performed to identify radiomic features predictive of nGBM prognosis.</p><p>Results: The distribution of the Gd-enhancing and T2-weighted image/fluid attenuated inversion recovery-high intensity lesions mainly occupied white matter. JCOG0911 consisted of more subjects with right-sided lesions. The median extent of resection of the Gd-enhancing lesions was 99%. The overall survival showed a nonsignificant negative trend with postoperative Gd-enhancing lesion volume (<i>P</i> = 0.22), with the hazard ratio increasing in parallel with its volume. The median PFS after registration was 302 and 308 days for local Response Evaluation Criteria in Solid Tumors (RECIST)-based and central RANO-based diagnoses. However, an apparent discrepancy was observed between the two in the early phase, presumably due to the misdiagnosis of pseudoprogression by local RECIST-based diagnosis. Radiomic analysis identified 28 radiomic features predictive of nGBM prognosis, 5 of which were those previously identified in a separate cohort. The constructed radiomics-based prognostic model stratified the cohort into high- and low-risk groups (<i>P</i> = 0.028).</p><p>Conclusion: Novel analytical methods that could be incorporated into future clinical trials were successfully tested. RANO and RECIST may not differ in progression calls if pseudoprogression is appropriately handled.</p>

DOI： 10.2463/mrms.mp.2024-0103

Web of Science

PubMed

CiNii Research

researchmap

DOI： 10.1093/neuonc/noae165.0803

Web of Science

DOI： 10.1093/neuonc/noae165.0748

Web of Science

DOI： 10.1093/neuonc/noae165.1270

Web of Science

DOI： 10.1093/neuonc/noae165.1103

Web of Science

DOI： 10.1093/neuonc/noae165.0672

Web of Science

Language：English   Publishing type：Research paper (scientific journal)  

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

Web of Science

Scopus

PubMed

researchmap

Language：English   Publisher：Nagoya Journal of Medical Science  

The anatomical characteristics of middle cerebral artery aneurysms make endovascular treatment difficult. This study evaluated the efficacy and safety of endovascular treatment of unruptured middle cerebral artery aneurysm in preventing rupture. A retrospective review of patients who underwent coil embolization for unruptured middle cerebral artery aneurysm between 2006 and 2022 at Nagoya University Hospital with at least 12 months followed up was conducted. Imaging and clinical outcomes were described using the Raymond classification and the modified Rankin Scale, respectively. Good imaging outcome was defined as complete occlusion or neck remnant and clinical outcome as modified Rankin Scale score of 0–2. Patients were divided into initial and recurrent group based on the number of treatments, pre- and post-stent groups based on when stents became available in Japan. A total of 77 patients (80 with aneurysms) were included in the final analysis. Their average age was 60.3 years, and their average follow-up period was 38 months. Favorable clinical outcomes were achieved for 96.2% among 66 (97.0%) initial and 11 (91.7%) recurrent aneurysms. Furthermore, good imaging outcomes were obtained in 90.0 %, and 5% had permanent symptomatic ischemic complications. The pre-stent group had a significantly higher proportion of patients with narrow-neck aneurysms than the post-stent group. There were no significant differences in terms of imaging and clinical outcomes or complication rates. The present study demonstrated that endovascular treatment of unruptured middle cerebral artery aneurysm was safe and effective in preventing rupture. The wide-neck aneurysm was also well embolized by using adjunctive technique.

DOI： 10.18999/nagjms.86.4.596

Web of Science

Scopus

PubMed

Language：English   Publisher：Nagoya Journal of Medical Science  

Selecting an appropriate microcatheter tip shape for paraclinoid aneurysms is difficult. Therefore, we devised an original simple and uniform three-dimensional (3D) spiral-shaping method of microcatheter and validated the characteristics and usefulness of this method for coil embolization of paraclinoid aneurysms using patient-specific silicone models. These silicone models were produced based on clinical data from four patients with four paraclinoid aneurysms that underwent endovascular treatment using the 3D spiral-shaping method. These models were classified into four types: superior, medial, inferior, and lateral corresponding to the aneurysm protrusion and locations (C3 or C2 segments by Fisher’s classification). Employing a pulsatile pump setup, two operators assessed the following items: navigation methods (pull and wire guiding), catheterization times, microcatheter tip position in the aneurysm, and the feasibility of inserting a framing coil by simple technique compared with three other shapes (straight, 90, pigtail). Three-dimensional spiral-shaped microcatheter could be placed in the medial and inferior type models of C3 segments and superior type model of C2 segment by the pullback method. Catheterization times using a 3D spiral-shaped catheter were significantly shorter than other shaped ones in the superior type models. No significant difference was found in another silicone model. Three-dimensional spiral- and pigtail-shaped catheters tended to position the tip at the center of the aneurysm. In conclusion, 3D spiral-shaped microcatheter was especially effective for the superior projected aneurysm at the C2 segment. The 3D spiral-shaping method can provide easy and secure navigation of the microcatheter into the paraclinoid aneurysms, ensuring optimal positioning for coil insertion.

DOI： 10.18999/nagjms.86.4.655

Web of Science

Scopus

PubMed

Language：English   Publishing type：Research paper (scientific journal)  

Mutations in the isocitrate dehydrogenase (IDH) gene are recognized as the key drivers in the oncogenesis of astrocytoma and oligodendroglioma. However, the significance of IDH mutation in tumor maintenance and malignant transformation has not been elucidated. We encountered a unique case of IDH-mutant astrocytoma that, upon malignant transformation, presented two distinct intratumoral components: one IDH-wildtype and one IDH-mutant. The IDH-wild-type component exhibited histological findings similar to those of small cell-type glioblastoma with a higher Ki-67 index than the IDH-mutant component. Despite their genetic divergence, both components exhibited similar comprehensive methylation profiles within the CpG island and were classified into methylation class of "Astrocytoma, IDH-mutant; High Grade" by the German Cancer Center (DKFZ) classifier v11.4. Phylogenetic analysis demonstrated that the IDH-wildtype component emerged as a subclonal component of the primary tumor. Detailed molecular analyses revealed that the loss of the IDH mutation was induced by the hemizygous loss of the entire arm of chromosome 2, on which IDH1 gene is located. Notably, the IDH-wild-type subclones uniquely acquired CDKN2A/B homozygous deletion and PDGFRA amplification, which is a marker of the aggressive phenotype of astrocytoma, IDH-mutant. Because these genetic abnormalities can drive oncogenic pathways, such as the PI3K/AKT/mTOR and RB signaling pathway, IDH-mutant gliomas that acquired these mutations were no longer dependent on the initial driver mutation, the IDH mutation. Molecular analysis of this unique case provides insight that in a subset of astrocytoma, IDH-mutant that acquired these genetic abnormalities, IDH mutation may not play a pivotal role in tumor growth and acquisition of these genetic abnormalities may contribute to the acquisition of resistance to IDH inhibitors.

DOI： 10.1186/s40478-024-01879-9

Open Access

Web of Science

Scopus

PubMed

researchmap

Language：English   Publisher：Journal of Neurosurgery: Case Lessons  

BACKGROUND High-grade astrocytoma with piloid features (HGAP) is a novel condition introduced in the 2021 World Health Organization classification. Given that it has been recently classified, reports clarifying its clinical features or diagnostic criteria are lacking, especially in cases of atypical presentation. Herein, the authors present a rare case of HGAP with repeated symptomatic hemorrhages. OBSERVATIONS A woman in her 20s presented with an acute headache and vertigo. Computed tomography and magnetic resonance imaging revealed a 2.5 × 2.8 × 2.3–cm hemorrhagic cerebellar mass with calcifications. After moderate improvement of her symptoms, she developed recurrent hemorrhage, and the tumor size increased (3.0 × 3.6 × 4.0 cm) 18 days later, necessitating resection. Pathological and molecular analyses confirmed the diagnosis of HGAP with an FGFR1-TACC1 fusion, MTAP/CDKN2A/B deletion, and SETD2 rearrangement. Radiologically, the presence of calcification and cystic components and the absence of perilesional edema were atypical features of previously reported HGAP. LESSONS Although recurrent symptomatic intracranial hemorrhages are rare in HGAP, enhancing lesions on magnetic resonance imaging suggest the need for resection to obtain tissue for molecular diagnosis and guide adjuvant treatment strategies.

DOI： 10.3171/CASE24395

Open Access

Web of Science

Scopus

PubMed

Language：English   Publishing type：Research paper (scientific journal)  

BACKGROUND: Treating meningeal tumours invading the large dural venous sinuses is a subject of debate regarding the approach for removing the intra-sinus components. Additionally, directly observing the invasion site of tumours invading the lateral wall of the sinus is difficult. METHOD: We describe our exo- and endoscopic two-step approach (EETA): an exoscope is used to remove the extra-sinus component, while an endoscope is used to observe the invaded lateral wall and remove the intra-sinus component. CONCLUSION: EETA can be a viable option for treating meningeal tumours invading the venous sinus owing to its high resection rate and low invasiveness.

DOI： 10.1007/s00701-024-06298-2

Open Access

Web of Science

Scopus

PubMed

researchmap

Language：English   Publishing type：Research paper (scientific journal)  

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

Web of Science

Scopus

PubMed

researchmap

Language：English   Publishing type：Research paper (scientific journal)  

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

Web of Science

Scopus

PubMed

researchmap

Language：English   Publishing type：Research paper (scientific journal)  

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

Open Access

Web of Science

Scopus

PubMed

researchmap

Language：English   Publishing type：Research paper (scientific journal)  

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

Web of Science

Scopus

PubMed

researchmap

DOI： 10.1136/ijgc-2024-IGCS.575

Web of Science

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

researchmap

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

researchmap

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

Open Access

Web of Science

Scopus

PubMed

Language：English   Publishing type：Research paper (scientific journal)  

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

Open Access

Web of Science

Scopus

PubMed

researchmap

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

Open Access

Web of Science

Scopus

PubMed

CiNii Research

Language：Japanese   Publisher：(一社)日本てんかん学会  

researchmap

Language：Japanese   Publisher：(一社)日本てんかん学会  

researchmap

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

researchmap

Language：English   Publisher：(一社)日本てんかん学会  

researchmap

Language：Japanese   Publisher：(一社)日本てんかん学会  

researchmap

Language：Japanese   Publisher：(一社)日本てんかん学会  

researchmap

Language：Japanese   Publisher：(一社)日本てんかん学会  

researchmap

Language：Japanese   Publisher：(一社)日本てんかん学会  

researchmap

Authorship：Last author   Language：English  

DOI： 10.7759/cureus.67884

Web of Science

PubMed

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

DOI： 10.1507/endocrine.100.s.hpt_19

CiNii Research

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

Open Access

Web of Science

Scopus

PubMed

Language：English   Publishing type：Research paper (scientific journal)  

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

Open Access

Web of Science

Scopus

PubMed

researchmap

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

DOI： 10.1007/s11060-024-04687-2

Web of Science

Scopus

PubMed

researchmap

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

Authorship：Last author   Language：English  

DOI： 10.7759/cureus.65547

Web of Science

PubMed

Language：English   Publishing type：Research paper (scientific journal)  

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

Web of Science

Scopus

PubMed

researchmap

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

DOI： 10.11477/mf.1436204969

PubMed

CiNii Research

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

Web of Science

Scopus

PubMed

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

Open Access

Web of Science

Scopus

PubMed

CiNii Research

Language：English   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

Open Access

Web of Science

Scopus

PubMed

Language：English   Publishing type：Research paper (scientific journal)  

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

Open Access

Web of Science

Scopus

PubMed

researchmap

Publisher：三輪書店  

DOI： 10.11477/mf.5002202293

CiNii Research

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

Web of Science

Scopus

PubMed

researchmap

Language：English   Publishing type：Research paper (scientific journal)  

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

Scopus

PubMed

researchmap

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

Web of Science

Scopus

PubMed

researchmap

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

researchmap

Language：English  

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

Web of Science

PubMed

researchmap

Language：English   Publishing type：Research paper (scientific journal)  

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

Open Access

Web of Science

Scopus

PubMed

researchmap

Language：English   Publishing type：Research paper (scientific journal)  

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

Web of Science

Scopus

PubMed

researchmap

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

researchmap

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

Open Access

Web of Science

Scopus

PubMed

CiNii Research

researchmap

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

Open Access

Web of Science

Scopus

PubMed

researchmap

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

Web of Science

Scopus

PubMed

researchmap

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

researchmap

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

Web of Science

Scopus

PubMed

researchmap

Language：English  

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

Web of Science

PubMed

researchmap

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

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

Web of Science

Scopus

PubMed

researchmap

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

Open Access

Web of Science

Scopus

PubMed

researchmap

Web of Science

Web of Science

Web of Science

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

Open Access

Web of Science

Scopus

PubMed

researchmap

Language：English   Publishing type：Research paper (scientific journal)  

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

Open Access

Web of Science

Scopus

PubMed

researchmap

Language：English   Publishing type：Research paper (scientific journal)  

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

Web of Science

Scopus

PubMed

researchmap

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

Web of Science

Scopus

PubMed

researchmap

Language：English   Publishing type：Research paper (scientific journal)  

DOI： 10.7759/cureus.52524

Web of Science

PubMed

researchmap

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

Web of Science

Scopus

PubMed

researchmap

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

Open Access

Web of Science

Scopus

PubMed

researchmap

Language：English   Publishing type：Research paper (scientific journal)  

DOI： 10.7759/cureus.51850

Web of Science

PubMed

researchmap

Language：English   Publishing type：Research paper (scientific journal)  

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

Open Access

Web of Science

Scopus

PubMed

researchmap

Language：English   Publishing type：Research paper (scientific journal)  

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

Web of Science

Scopus

PubMed

researchmap

Language：English   Publishing type：Research paper (scientific journal)  

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

Web of Science

Scopus

PubMed

researchmap

Language：Japanese   Publisher：The Japanese Society of Spinal Surgery  

DOI： 10.2531/spinalsurg.38.71

CiNii Research

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

PubMed

CiNii Research

researchmap

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

Open Access

Scopus

PubMed

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

CiNii Research

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

CiNii Research

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

CiNii Research

researchmap

Language：Japanese   Publisher：The Japanese Society of Pediatric Hematology / Oncology  

<p>The authors present a case of ependymoma, which is a rare surgical disease. To determine the significance of ependymoma surgery, we evaluated the frequency of repeat ependymoma surgeries proportionate to that of 235 neuroepithelial tumors/341 surgeries performed by the first author between 1997 and 2023. Repeat ependymoma surgeries accounted for 10/92 (11%) cases with WHO grades 2 and 3 (32 of 136 repeat surgeries [24%]). Ependymoma surgery to achieve local tumor control is of great significance. The treatment of pediatric/adolescent and young adult brain tumors aims to prolong the prognosis and reduce the treatment side effects. Therefore, neurosurgeons must maximize tumor resection while minimizing surgical morbidity, if surgery contributes to local tumor control.</p>

DOI： 10.11412/jspho.61.238

CiNii Research

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

<p>Clazosentan is used to suppress the onset of cerebral vasospasm, delayed cerebral ischemia (DCI), and new cerebral infarctions after subarachnoid hemorrhage. Here we retrospectively examine our initial experience using this drug in our department. Evaluation items included patient background factors, clinical characteristics, spasms on imaging, DCI/new cerebral infarction, complications associated with fluid retention, and other complications. Thirty-two patients were included; among them, clazosentan was administered to 25. In the clazosentan group, 10 patients (31%) exhibited spasms on imaging, one (4%) experienced DCI, and four (16%) had a new cerebral infarction. Complications associated with fluid retention occurred in 13 patients (52%). Although differences were observed in the phase III study in patient background factors, clinical characteristics, and concomitant medications, clazosentan effectively prevented DCI and new cerebral infarctions. Additional cases should be examined to establish an ideal body fluid management protocol.</p>

DOI： 10.2335/scs.52.258

CiNii Research

Language：English   Publishing type：Research paper (scientific journal)  

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

Web of Science

Scopus

PubMed

researchmap

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

Open Access

Web of Science

Scopus

PubMed

CiNii Research

researchmap

Language：English   Publishing type：Research paper (scientific journal)  

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

Web of Science

Scopus

PubMed

researchmap

Language：English   Publishing type：Research paper (scientific journal)  

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

Web of Science

Scopus

PubMed

researchmap

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

DOI： 10.1007/s10143-023-02196-w

Web of Science

Scopus

PubMed

researchmap

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

Language：English   Publishing type：Research paper (scientific journal)  

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

Web of Science

Scopus

PubMed

researchmap

Publishing type：Research paper (other academic)  

Web of Science

researchmap

Publishing type：Research paper (other academic)  

researchmap

researchmap

researchmap

researchmap

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

DOI： 10.1016/j.seizure.2023.09.018

Web of Science

Scopus

PubMed

researchmap

Language：English   Publishing type：Research paper (scientific journal)  

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

Open Access

Web of Science

Scopus

PubMed

researchmap

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

Open Access

Web of Science

Scopus

PubMed

CiNii Research

researchmap

Web of Science

Web of Science

Web of Science

Web of Science

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

DOI： 10.1038/s41598-023-45755-3

Open Access

Web of Science

Scopus

PubMed

researchmap

Language：English   Publishing type：Research paper (scientific journal)   Publisher：Scientific Reports  

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

Open Access

Web of Science

Scopus

PubMed

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

DOI： 10.1016/j.wneu.2023.07.038

Web of Science

Scopus

PubMed

researchmap

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

DOI： 10.1016/j.seizure.2023.07.007

Web of Science

Scopus

PubMed

researchmap

Language：English   Publishing type：Research paper (scientific journal)  

DOI： 10.1016/j.wneu.2023.07.002

Web of Science

Scopus

PubMed

researchmap

Language：English   Publishing type：Research paper (scientific journal)  

OBJECTIVE: Cerebral infarction is a common complication in patients undergoing revascularization surgery for moyamoya disease (MMD). Although previous statistical evaluations have identified several risk factors for postoperative brain ischemia, the ability to predict its occurrence based on these limited predictors remains inadequately explored. This study aimed to assess the feasibility of machine learning algorithms for predicting cerebral infarction after revascularization surgery in patients with MMD. METHODS: This retrospective study was conducted across two centers and harnessed data from 512 patients with MMD who had undergone revascularization surgery. The patient cohort was partitioned into internal and external datasets. Using perioperative clinical data from the internal cohort, three distinct machine learning algorithms-namely the support vector machine, random forest, and light gradient-boosting machine models-were trained and cross-validated to predict the occurrence of postoperative cerebral infarction. Predictive performance validity was subsequently assessed using an external dataset. Shapley additive explanations (SHAP) analysis was conducted to augment the prediction model's transparency and to quantify the impact of each input variable on shaping both the aggregate and individual patient predictions. RESULTS: In the cohort of 512 patients, 33 (6.4%) experienced postrevascularization cerebral infarction. The cross-validation outcomes revealed that, among the three models, the support vector machine model achieved the largest area under the receiver operating characteristic curve (ROC-AUC) at mean ± SD 0.785 ± 0.052. Notably, during external validation, the light gradient-boosting machine model exhibited the highest accuracy at 0.903 and the largest ROC-AUC at 0.710. The top-performing prediction model utilized five input variables: postoperative serum gamma-glutamyl transpeptidase value, positive posterior cerebral artery (PCA) involvement on preoperative MRA, infarction as the rationale for surgery, presence of an infarction scar on preoperative MRI, and preoperative modified Rankin Scale score. Furthermore, the SHAP analysis identified presence of PCA involvement, infarction as the rationale for surgery, and presence of an infarction scar on preoperative MRI as positive influences on postoperative cerebral infarction. CONCLUSIONS: This study indicates the usefulness of employing machine learning techniques with routine perioperative data to predict the occurrence of cerebral infarction after revascularization procedures in patients with MMD.

DOI： 10.3171/2024.1.JNS232173

Web of Science

Scopus

PubMed

researchmap

Language：English   Publishing type：Research paper (scientific journal)  

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

Open Access

Web of Science

Scopus

PubMed

researchmap

Language：English   Publishing type：Research paper (scientific journal)  

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

Open Access

Web of Science

Scopus

PubMed

researchmap

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

DOI： 10.1111/jnp.12345

Web of Science

Scopus

PubMed

researchmap

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

PubMed

CiNii Research

researchmap

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

researchmap

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

DOI： 10.1507/endocrine.99.s.hpt_4

CiNii Research

Publisher：医歯薬出版  

DOI： 10.32118/ayu28609707

CiNii Research

researchmap

Language：English   Publishing type：Research paper (scientific journal)  

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

Open Access

Web of Science

Scopus

PubMed

researchmap

Language：English   Publishing type：Research paper (scientific journal)  

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

Web of Science

Scopus

PubMed

researchmap

Language：English   Publishing type：Research paper (scientific journal)  

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

Web of Science

Scopus

PubMed

researchmap

Language：English   Publishing type：Research paper (scientific journal)  

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

Web of Science

Scopus

PubMed

researchmap

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

Open Access

Web of Science

Scopus

PubMed

CiNii Research

researchmap

Language：English   Publishing type：Research paper (scientific journal)  

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

Web of Science

Scopus

PubMed

researchmap

Authorship：Last author   Language：English  

DOI： 10.7759/cureus.42510

Web of Science

PubMed

researchmap

Language：English   Publishing type：Research paper (scientific journal)  

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

Web of Science

Scopus

PubMed

researchmap

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

CiNii Research

researchmap

Language：English   Publishing type：Research paper (scientific journal)  

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

Web of Science

PubMed

researchmap

Authorship：Last author  

DOI： 10.1161/SVIN.122.000759

Web of Science

researchmap

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

researchmap

Language：English   Publishing type：Research paper (scientific journal)  

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.

DOI： 10.1038/s41598-023-37834-2

Open Access

Web of Science

Scopus

PubMed

researchmap

Language：English   Publishing type：Research paper (scientific journal)  

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

Open Access

Web of Science

Scopus

PubMed

researchmap

Language：English   Publishing type：Research paper (scientific journal)  

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

Open Access

Web of Science

Scopus

PubMed

researchmap

Language：English   Publishing type：Research paper (scientific journal)  

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

Web of Science

Scopus

PubMed

researchmap

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

Open Access

Web of Science

Scopus

PubMed

CiNii Research

researchmap

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

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

Open Access

Web of Science

Scopus

PubMed

CiNii Research

researchmap

Authorship：Last author   Language：Japanese   Publishing type：Research paper (scientific journal)  

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

PubMed

CiNii Research

researchmap

Publishing type：Research paper (scientific journal)  

DOI： 10.2176/jns-nmc.er.2021-0390

Open Access

Web of Science

Scopus

researchmap

Language：English  

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

Web of Science

Scopus

PubMed

researchmap

Language：English   Publishing type：Research paper (scientific journal)  

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

Web of Science

Scopus

PubMed

researchmap

Language：English   Publishing type：Research paper (scientific journal)  

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

Web of Science

Scopus

PubMed

researchmap

Language：English   Publishing type：Research paper (scientific journal)  

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

Web of Science

Scopus

PubMed

researchmap

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

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

Open Access

Web of Science

Scopus

PubMed

CiNii Research

researchmap

Language：English   Publishing type：Research paper (scientific journal)  

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

Web of Science

Scopus

PubMed

researchmap

Language：English   Publishing type：Research paper (scientific journal)  

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

Web of Science

Scopus

PubMed

researchmap

Language：English   Publishing type：Research paper (scientific journal)  

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

Web of Science

Scopus

PubMed

researchmap

Language：English   Publishing type：Research paper (scientific journal)   Publisher：Oxford University Press (OUP)  

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

Open Access

Web of Science

Scopus

PubMed

researchmap

Language：English   Publishing type：Research paper (scientific journal)  

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

Open Access

Web of Science

Scopus

PubMed

researchmap

Language：English   Publishing type：Research paper (scientific journal)  

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

Web of Science

Scopus

PubMed

researchmap

Language：English   Publishing type：Research paper (scientific journal)  

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

Web of Science

Scopus

PubMed

researchmap

Language：English   Publishing type：Research paper (scientific journal)  

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.

DOI： 10.1007/s00701-022-05415-3

Web of Science

Scopus

PubMed

researchmap

Language：English  

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

Web of Science

PubMed

researchmap

Language：English   Publishing type：Research paper (scientific journal)  

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

Web of Science

Scopus

PubMed

researchmap

Language：English   Publishing type：Research paper (scientific journal)  

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

Open Access

Web of Science

Scopus

PubMed

researchmap

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

researchmap

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

researchmap

Language：English   Publishing type：Research paper (scientific journal)  

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

Open Access

Web of Science

Scopus

PubMed

researchmap

Language：English  

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

Web of Science

PubMed

researchmap

Web of Science

researchmap

researchmap

Language：English  

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

Web of Science

Scopus

PubMed

researchmap

Language：English   Publishing type：Research paper (scientific journal)  

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

Web of Science

Scopus

PubMed

researchmap

Language：English   Publishing type：Research paper (scientific journal)  

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

Web of Science

Scopus

PubMed

researchmap

researchmap

Language：English   Publishing type：Research paper (scientific journal)  

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

Web of Science

Scopus

PubMed

researchmap

researchmap

researchmap

Web of Science

Web of Science

Web of Science

Web of Science

Language：English   Publishing type：Research paper (scientific journal)  

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

PubMed

researchmap

Language：English   Publishing type：Research paper (scientific journal)  

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

Open Access

Web of Science

Scopus

PubMed

researchmap

Language：English   Publishing type：Research paper (scientific journal)  

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

Web of Science

Scopus

PubMed

researchmap

Language：English   Publishing type：Research paper (scientific journal)  

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

Web of Science

Scopus

PubMed

researchmap

Authorship：Last author   Language：Japanese   Publishing type：Research paper (scientific journal)  

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

PubMed

CiNii Research

researchmap

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

researchmap

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

CiNii Research

researchmap

Publishing type：Research paper (scientific journal)   Publisher：Elsevier BV  

DOI： 10.1016/j.abst.2023.09.001

researchmap

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

<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

Web of Science

PubMed

CiNii Research

researchmap

Authorship：Lead author,　Last author,　Corresponding author   Language：Japanese   Publisher：The Japanese Congress of Neurological Surgeons  

<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

Open Access

Scopus

CiNii Research

researchmap

Language：English  

BACKGROUND: An arteriovenous fistula (AVF) at the craniocervical junction (CCJ) is a rare vascular malformation. Definitive diagnosis and curative treatment of CCJ AVF are challenging. CASE DESCRIPTION: A 77-year-old man presented with subarachnoid hemorrhage. Cerebral angiography showed an AVF at the CCJ, which drained into a radicular vein. The lesion was fed by a vertebral artery, anterior and lateral spinal arteries (LSAs), and the occipital artery (OA). There were two unique structures: the LSA originating from the posterior inferior cerebellar artery of the extracranial V3 segment and the OA feeding the shunt. Curative treatment involved two steps: endovascular embolization of feeders using Onyx and surgical shunt disconnection. Feeding arteries were blackened by Onyx, which helped identify the location of the shunt. The shunt was located behind the first cervical (C1) spinal nerve, and the draining vein was confirmed on the deep side of the nerve. A clip was applied to the draining vein distal to the shunt. Tiny vessels supplying the shunt were then coagulated referring to blackened arteries. CONCLUSION: A radicular AVF at the CCJ along the C1 spinal nerve had unique vascular structures. Definitive diagnosis and curative treatment were achieved by combining endovascular embolization using Onyx and direct surgery.

DOI： 10.25259/SNI_122_2023

Open Access

Scopus

PubMed

researchmap

Language：English   Publisher：The Japan Neurosurgical Society  

Hereditary neuropathy with liability to pressure palsies is an extremely rare genetic disorder; it is an autosomal dominant disorder with a high incidence of neuropathic and/or musculoskeletal pain. A case of achieving pain relief by spinal cord stimulation using differential target multiplexed stimulation for a 44-year-old female patient with hereditary neuropathy with liability to pressure palsies who was experiencing severe pain in her back, face, and all four limbs is presented. In her early teens, the initial symptoms were numbness and weakness of a limb after movement, which improved spontaneously. Transient pain in her back followed by systemic and persistent muscle weakness and pain developed. Deletion of the gene for peripheral myelin protein 22 was detected by peripheral nerve biopsy. The diagnosis of hereditary neuropathy with liability to pressure palsies was made in her early thirties. A spinal cord stimulation trial was performed because her severe pain continued despite administering many medications. Therefore, two spinal cord stimulation systems were implanted at the C3-5 and Th8-9 levels by two procedures. Pain in her back, arms, and legs decreased from 8 to 1, 5 to 1, and 6 to 2 on the numerical rating scale, respectively. Furthermore, opioid usage was tapered. The pain of hereditary neuropathy with liability to pressure palsies has a complicated pathogenesis and is resistant to pharmacological treatment. Spinal cord stimulation using differential target multiplexed stimulation may be a viable treatment option.

DOI： 10.2176/jns-nmc.2023-0023

PubMed

CiNii Research

researchmap

Language：English   Publisher：The Japan Neurosurgical Society  

Central poststroke pain is a chronic, intractable, central neuropathic pain. Spinal cord stimulation is a neuromodulation therapy for chronic neuropathic pain. The conventional stimulation method induces a sense of paresthesia. Fast-acting subperception therapy is one of the latest new stimulation methods without paresthesia. A case of achieving pain relief of central poststroke pain affecting both the arm and leg on one side by double-independent dual-lead spinal cord stimulation using fast-acting subperception therapy stimulation is presented. A 67-year-old woman had central poststroke pain due to a right thalamic hemorrhage. The numerical rating scale scores of the left arm and leg were 6 and 7, respectively. Using dual-lead stimulation at the Th 9-11 levels, a spinal cord stimulation trial was performed. Fast-acting subperception therapy stimulation achieved pain reduction in the left leg from 7 to 3. Therefore, a pulse generator was implanted, and the pain relief continued for 6 months. Then, two additional leads were implanted at the C 3-5 levels, and pain in the arm decreased from 6 to 4. Independent setting and adjustments of the dual-lead stimulation were required because the thresholds of paresthesia perception were significantly different. To achieve pain relief in both the arm and leg, double-independent dual-lead stimulation placed at cervical and thoracic levels is an effective treatment. Fast-acting subperception therapy stimulation may be effective for central poststroke pain, especially in cases where the paresthesia is perceived as uncomfortable or the conventional stimulation itself is ineffective.

DOI： 10.2176/jns-nmc.2022-0336

PubMed

CiNii Research

researchmap

Language：English   Publisher：The Japan Neurosurgical Society  

A 68-year-old man presented with a Jefferson fracture leading to lower cranial nerve palsies affecting the ninth, tenth, and twelfth cranial nerves with a traumatic basilar impression. On the X day, the patient underwent occipitocervical posterior fixation surgery; the surgery was uneventful. However, just after the surgery, epipharyngeal palsy and airway obstruction occurred. Consequently, tracheostomy was needed. On the X+8 day, speech-language pathology (SLP) therapy was initiated for decannulation. On the X+21 day, the patient could clear all the checkpoints and was decannulated. On the X+36 day, the patient was discharged home and SLP therapy was continued. On the X+171 day, his SLP therapy was halted. However, the patient continued to complain that he could not speak as fast as before, and his quality of life remained compromised. Some studies reported that lower cranial nerve palsies affecting the ninth to the twelfth cranial nerve occur in conjunction with Jefferson fractures. Thus, SLP therapy is crucial for Jefferson fracture cases.

DOI： 10.2176/jns-nmc.2022-0060

PubMed

CiNii Research

researchmap

Language：English   Publisher：The Japan Neurosurgical Society  

A 61-year-old man presented with massive epistaxis, amaurosis, nausea, and severe headache. A detailed examination revealed a subarachnoid hemorrhage and prolactinoma. Angiography showed a small internal carotid artery pseudoaneurysm and inadequate collateral circulation; thus, uncomplicated coil embolization was performed. Considering the side effects of medication, such as cerebrospinal fluid rhinorrhea, the patient was followed up for asymptomatic prolactinoma without medication after discharge. At 40 months later, aneurysm recurrence was confirmed. Flow diverter device placement was performed, and the outcomes were excellent. In the present report, we described a rare case of a ruptured internal carotid artery aneurysm in an untreated prolactinoma and discussed the literature.

DOI： 10.2176/jns-nmc.2022-0355

PubMed

CiNii Research

researchmap

Language：English   Publishing type：Research paper (scientific journal)  

DOI： 10.25259/SNI_606_2023

Open Access

Scopus

PubMed

researchmap

Language：English   Publisher：The Japan Neurosurgical Society  

Intracranial aneurysms (IA) in infants are reportedly rare at 0.5% to 4.5% of all aneurysms. Furthermore, subarachnoid hemorrhage in infants younger than three months are even rarer as it has been reported in approximately 20 cases only till date. A 3-month-old infant with seizures and impaired consciousness was admitted to our hospital. Three-dimensional computed tomography angiography (3D-CTA) revealed a dissecting aneurysm with a maximum diameter of 13 mm in the right M2. Internal trapping using detachable coil were successfully performed, following which he was discharged without significant neurological deficit after one month of onset. Thus, we have reported a rare case of a large ruptured dissecting IA in a 3-month-old infant, in the right middle cerebral artery (MCA), successfully treated with an endovascular therapy, along with a literature review.

DOI： 10.2176/jns-nmc.2022-0369

PubMed

CiNii Research

researchmap

Language：English  

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

Open Access

Web of Science

Scopus

PubMed

researchmap

Language：English   Publishing type：Research paper (scientific journal)  

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

Open Access

Web of Science

Scopus

PubMed

researchmap

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

researchmap

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

researchmap

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

Language：English   Publishing type：Research paper (scientific journal)  

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

Web of Science

Scopus

PubMed

researchmap

Language：English   Publishing type：Research paper (scientific journal)  

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

Web of Science

Scopus

PubMed

researchmap

researchmap

Web of Science

Language：English   Publishing type：Research paper (scientific journal)  

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

Web of Science

Scopus

PubMed

researchmap

Web of Science

Web of Science

Language：English  

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

Web of Science

Scopus

PubMed

researchmap

researchmap

researchmap

Language：English   Publishing type：Research paper (scientific journal)  

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

PubMed

researchmap

Language：English   Publishing type：Research paper (scientific journal)  

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

Web of Science

Scopus

PubMed

researchmap

Language：English   Publishing type：Research paper (scientific journal)  

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

Web of Science

Scopus

PubMed

researchmap

Language：English   Publishing type：Research paper (scientific journal)  

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

Web of Science

Scopus

PubMed

researchmap

Language：English   Publishing type：Research paper (scientific journal)  

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

Open Access

Web of Science

Scopus

PubMed

researchmap

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

researchmap

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

researchmap

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

researchmap

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

researchmap

Language：English   Publishing type：Research paper (scientific journal)  

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

Web of Science

Scopus

PubMed

researchmap

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

researchmap

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

researchmap

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

researchmap

Language：English  

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

Web of Science

PubMed

researchmap

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

DOI： 10.1507/endocrine.98.s.hpt_25

CiNii Research

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

DOI： 10.1507/endocrine.98.s.hpt_72

CiNii Research

researchmap