Language：English   Publisher：The Japan Neurosurgical Society  

<p>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.</p>

DOI： 10.2176/jns-nmc.2022-0271

PubMed

CiNii Research

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

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Language：English   Publisher：Brain Tumor Pathology  

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

DOI： 10.1007/s10014-023-00459-4

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Language：English   Publisher：Brain Tumor Pathology  

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

DOI： 10.1007/s10014-023-00458-5

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Language：English  

DOI： 10.3171/CASE22563

PubMed

Language：English  

DOI： 10.3171/2023.1.PEDS22345

PubMed

Language：English   Publisher：Frontiers in Endocrinology  

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

DOI： 10.3389/fendo.2023.1130465

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Language：English   Publisher：Journal of Neurosurgery  

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

DOI： 10.3171/2022.5.JNS22579

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PubMed

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

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Language：English   Publisher：The Japan Neurosurgical Society  

<p>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.</p>

DOI： 10.2176/jns-nmc.2021-0390

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CiNii Research

Language：English   Publisher：Journal of Neurosurgery  

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

DOI： 10.3171/2022.6.JNS22815

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DOI： 10.1002/ccr3.7086

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PubMed

Language：English   Publisher：World Neurosurgery  

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

DOI： 10.1016/j.wneu.2022.12.053

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Language：English   Publisher：Cancer Medicine  

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

DOI： 10.1002/cam4.5512

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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

PubMed

Language：English   Publisher：Nagoya Journal of Medical Science  

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

DOI： 10.18999/nagjms.85.1.127

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Language：English   Publisher：Nagoya Journal of Medical Science  

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

DOI： 10.18999/nagjms.85.1.50

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DOI： 10.7759/cureus.34672

PubMed

Language：English   Publisher：Clinical Neurology and Neurosurgery  

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

DOI： 10.1016/j.clineuro.2022.107583

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Language：English   Publisher：British Journal of Neurosurgery  

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

DOI： 10.1080/02688697.2023.2170328

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Language：English   Publisher：Scientific Reports  

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

DOI： 10.1038/s41598-022-25928-2

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Language：English   Publisher：Acta Neurochirurgica  

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

DOI： 10.1007/s00701-023-05489-7

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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

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Language：English   Publisher：Journal of Neurosurgery  

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.

DOI： 10.3171/2022.3.JNS212460

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Language：Japanese   Publisher：株式会社医学書院  

DOI： 10.11477/mf.1436204711

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CiNii Research

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

Language：English   Publisher：The Japan Neurosurgical Society  

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

DOI： 10.2176/jns-nmc.2022-0055

CiNii Research

Language：English   Publisher：The Japan Neurosurgical Society  

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

DOI： 10.2176/jns-nmc.2022-0056

CiNii Research

Language：English   Publisher：The Japan Neurosurgical Society  

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

DOI： 10.2176/jns-nmc.2022-0036

CiNii Research

Language：English   Publisher：Interventional Neuroradiology  

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

DOI： 10.1177/15910199221145526

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Language：English   Publisher：Frontiers in Human Neuroscience  

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

DOI： 10.3389/fnhum.2022.1065459

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Language：English   Publisher：Epilepsia Open  

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

DOI： 10.1002/epi4.12634

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

Language：English   Publisher：Acta Neurochirurgica  

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

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Language：English   Publisher：World Neurosurgery  

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

DOI： 10.1016/J.WNEU.2022.08.116

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Language：English   Publisher：World Neurosurgery  

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

DOI： 10.1016/j.wneu.2022.08.147

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Language：English   Publisher：Nagoya Journal of Medical Science  

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

DOI： 10.18999/nagjms.84.4.762

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Language：English   Publisher：Nagoya Journal of Medical Science  

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

DOI： 10.18999/nagjms.84.4.884

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Language：English   Publisher：Neurosurgical Review  

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

DOI： 10.1007/s10143-022-01820-5

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Language：English   Publisher：Acta Neurochirurgica  

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

DOI： 10.1007/s00701-022-05273-z

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Language：Japanese   Publisher：Journal of Neural Transmission  

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

DOI： 10.1007/s00702-022-02528-y

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Language：English   Publisher：American Journal of Neuroradiology  

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

DOI： 10.3174/ajnr.A7646

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

Language：English   Publisher：Journal of Neurosurgery: Pediatrics  

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

DOI： 10.3171/2022.6.PEDS2241

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Language：English  

DOI： 10.7759/cureus.28313

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

DOI： 10.1507/endocrine.98.s.hpt_25

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

Language：English  

DOI： 10.3171/CASE22256

PubMed

Language：English   Publisher：Nagoya Journal of Medical Science  

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

DOI： 10.18999/nagjms.84.3.678

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Language：English   Publisher：World Neurosurgery  

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

DOI： 10.1016/j.wneu.2022.04.079

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

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

DOI： 10.3171/2021.7.JNS21990

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Language：English   Publisher：Journal of Stroke and Cerebrovascular Diseases  

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

DOI： 10.1016/j.jstrokecerebrovasdis.2022.106526

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Publisher：三輪書店  

DOI： 10.11477/mf.5002201799

CiNii Research

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

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PubMed

Language：Japanese   Publisher：BMC Neurology  

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

DOI： 10.1186/s12883-022-02749-4

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PubMed

Language：English   Publisher：LIPPINCOTT WILLIAMS & WILKINS  

Web of Science

Language：Japanese   Publisher：Interventional Neuroradiology  

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

DOI： 10.1177/15910199211031765

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PubMed

Language：Japanese   Publisher：Neurosurgical Review  

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

DOI： 10.1007/s10143-022-01774-8

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Language：Japanese   Publisher：Acta Neurochirurgica  

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

DOI： 10.1007/s00701-022-05227-5

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PubMed

Language：Japanese   Publisher：Neurospine  

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

DOI： 10.14245/ns.2244168.084

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Language：Japanese   Publisher：Frontiers in Neurology  

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

DOI： 10.3389/fneur.2022.874826

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Language：Japanese   Publisher：Scientific Reports  

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

DOI： 10.1038/s41598-022-11321-6

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Language：Japanese   Publisher：Neurosurgical Review  

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

DOI： 10.1007/s10143-021-01685-0

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PubMed

Language：Japanese   Publisher：Neurosurgical Review  

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

DOI： 10.1007/s10143-021-01686-z

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PubMed

Language：Japanese   Publisher：Neurosurgical Review  

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

DOI： 10.1007/s10143-021-01634-x

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PubMed

Language：Japanese   Publisher：The Japan Neurosurgical Society  

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

DOI： 10.2176/jns-nmc.2021-0355

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PubMed

Language：Japanese   Publisher：World Neurosurgery  

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

DOI： 10.1016/j.wneu.2021.12.115

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Publisher：三輪書店  

DOI： 10.11477/mf.5002201765

CiNii Research

Language：Japanese   Publisher：Journal of Clinical Endocrinology and Metabolism  

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

DOI： 10.1210/clinem/dgab829

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PubMed

Language：English  

DOI： 10.3171/CASE21704

PubMed

Language：English  

DOI： 10.3171/CASE21673

PubMed

Language：English   Publisher：Journal of Neurosurgery  

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

DOI： 10.3171/2022.5.JNS22411

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Language：English  

DOI： 10.3171/CASE21628

PubMed

Language：Japanese   Publisher：株式会社医学書院  

DOI： 10.11477/mf.1436204529

PubMed

CiNii Research

Language：Japanese   Publisher：The Japan Neurosurgical Society  

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

DOI： 10.2176/nmc.oa.2021-0173

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PubMed

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

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

DOI： 10.1007/s10014-021-00418-x

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Language：Japanese   Publisher：The Japan Stroke Society  

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

DOI： 10.3995/jstroke.11028

CiNii Research

Language：English   Publisher：The Japanese Society for Neuroendovascular Therapy  

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

DOI： 10.5797/jnet.cr.2021-0029

CiNii Research

Language：English   Publisher：The Japanese Society for Neuroendovascular Therapy  

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

DOI： 10.5797/jnet.cr.2021-0095

CiNii Research

Language：Japanese   Publisher：The Japanese Society of Spinal Surgery  

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

DOI： 10.2531/spinalsurg.36.88

CiNii Research

Language：English  

DOI： 10.25259/SNI_453_2022

PubMed

Language：English   Publisher：Surgical Neurology International  

Background: The aim of this study was to investigate the association between early postoperative slow flow in bypass grafts and preoperative focal cerebral blood flow (CBF) in patients who underwent combined surgery for moyamoya disease (MMD). Methods: The subjects were 18 patients (22 surgeries) who underwent single photon emission computed tomography (SPECT) before surgery. The CBF value of the middle cerebral artery territory was extracted from the SPECT data, and the value relative to the ipsilateral cerebellar CBF (relative CBF, or RCBF) was calculated. The association between RCBF and early postoperative slow flow in the bypass graft was investigated. In addition, the correlation between the revascularization effect and preoperative RCBF was analyzed. Results: In four of 22 surgeries (18.2%), slow flow in the bypass graft was identified in the early postoperative period. Preoperative RCBF in the slow flow and patent groups was 0.86 ± 0.15 and 0.87 ± 0.15, respectively, with no significant difference (P = 0.72). The signal intensity of four slow-flowed bypasses was improved in all cases on magnetic resonance angiography images captured during the chronic phase (mean of 3.3 months postoperatively). The revascularization scores were 2 ± 0.82 and 2.1 ± 0.68 in the slow flow and patent groups, respectively, and did not differ significantly (P = 0.78). A significant correlation was not observed between preoperative RCBF and the revascularization effect. Conclusion: No significant association was observed between preoperative RCBF and early postoperative slow flow in bypass grafts in patients with MMD undergoing combined surgery. Given the high rate of improved depiction of slow-flowed bypass in the chronic postoperative phase, the conceptual significance of an opportune surgical intervention is to maintain CBF by supporting the patient's own intracranial-extracranial conversion function.

DOI： 10.25259/SNI_772_2022

Scopus

PubMed

Language：English   Publisher：Surgical Neurology International  

Background: A downward penetrating endplate screw (PES) technique combined with caudal anchor screws inserted in the upward direction under O-arm navigation (i.e., crossing screw technique) avoided screw backout and proximal junctional kyphosis (PJK) in three patients with osteoporotic vertebral body fractures and diffuse idiopathic skeletal hyperostosis (DISH). Methods: The PES techniques were utilized for patients with T12 (one patient) and L1 (two patients) spontaneous fusion across the targeted vertebrae, with minimal damage to the involved endplates/intervertebral discs. The average number of instrumented vertebrae was 5.3. Results: There were no perioperative complications over the mean follow-up period of 28.7 months; no screw loosening, and no PJK. Conclusion: The PES technique prevented screw backout, and PJK in three patients with lumbar osteoporotic vertebral fractures and DISH.

DOI： 10.25259/SNI_762_2022

Scopus

PubMed

Language：English   Publisher：Surgical Neurology International  

Background: The optimal treatment of lumbar canal stenosis (LCS) associated with osteoporotic vertebral fractures (OVFs) remains unclear. Here, we have combined a minimally invasive unilateral laminotomy for bilateral decompression (ULBD) alone with balloon kyphoplasty (BKP) for LCS aggravated by OVF. Methods: ULBD with BKP was performed in three patients who showed LCS associated with OVFs on MR images with progressive lower extremity neurological deficits. Clinical outcomes were assessed using the numerical rating scale (NRS) and the Japanese Orthopaedic Association (JOA) score. Radiological outcomes were evaluated using multiple parameters (i.e., fractured vertebral body height, lumbar lordosis [LL], and focal angle of the fractured vertebral body). Results: Over 6 postoperative months, the NRS and JOA scores were clearly improved while radiological parameters remained maintained (i.e., loss of fractured vertebral body height was only 0.3-1.4 mm in all cases). Two of the three cases showed restoration of LL and focal angle postoperatively. Conclusion: The combination of ULBD with BKP is an effective option for LCS aggravated by OVF.

DOI： 10.25259/SNI_971_2022

Scopus

PubMed

Language：Japanese   Publisher：The Japanese Society for Pediatric Neurosurgery  

<p>Scurvy due to Vit.C deficiency was widely prevalent in the world during the Age of Discovery. It reduces strength of vessel wall and causes bleeding. Nowadays, it is a rare and uncommon disease. We present a case of epidural hematoma associated with scurvy.</p><p> A 4-year-old girl admitted to our hospital with symptoms of difficulty in walking, leg pain, and bleeding from the swelled gums lasting 2.5 months. The blood cell count, hemogram and coagulation factors in laboratory data were normal. She had a headache without obvious trauma. CT scanning revealed an epidural hematoma in the right frontal region. A removal of epidural hematoma with hemostasis was performed. Re-bleeding did not occur after surgery, but the anemia was progressed. Other symptoms had also gotten worse. An interview about her daily meal revealed she was extremely picky eater. As the serum Vit.C concentration was quite low, she was finally diagnosed with scurvy. After starting oral administration of Vit.C, her symptoms quickly improved.</p><p> In modern times, it is extremely rare to experience nutritional disorders in developed country. However, mal-nutrition can be occur due to some reason like this case. Intracranial hemorrhage without history of underlying disease or trauma and accompanied with multi-organ findings may be caused by nutritional disorders.</p>

DOI： 10.34544/jspn.47.3_309

CiNii Research

Language：Japanese   Publisher：The Japanese Society of Spinal Surgery  

<p>  The clinical applications of three-dimensional (3D) printing in the field of spine surgery and education have recently begun to progress. Technological advances have made it relatively inexpensive and easy to create 3D models. Herein, we explain how to construct a full-scale model using a 3D printer, and review articles on the current clinical applications and limitations. Creation of a 3D model based on the patient’s radiographic image data facilitates understanding of the complicated anatomy. This model enables improvements in visual understanding, preoperative surgical planning, and gives the surgeon a training opportunity. Furthermore, 3D printer technology can also be useful during surgery, and 3D printed surgical guidance systems have previously been successfully used for spine surgery. 3D printing patient-specific guides and templates are highly useful to facilitate safe screw insertion. In addition, 3D printers can be used to create patient-specific implants.</p>

DOI： 10.2531/spinalsurg.36.296

CiNii Research

Language：Japanese   Publisher：日本神経治療学会  

DOI： 10.15082/jsnt.39.4_655

CiNii Research

Language：Japanese   Publisher：Neurospine  

Objective: The purpose of this study is to find the clinical and radiographic characteristics of traumatic craniocervical junction (CCJ) injuries requiring occipitocervical fusion (OC fusion) for early diagnosis and surgical intervention. Methods: We retrospectively reviewed 12 patients with CCJ injuries presenting to St. Mi-chaels Hospital in Toronto who underwent OC fusion and looked into the following vari-ables; (1) initial trauma data on emergency room arrival, (2) associated injuries, (3) imaging characteristics of computed tomography (CT) scan and magnetic resonance imaging (MRI), (4) surgical procedures, surgical complications, and neurological outcome. Results: All patients were treated as acute spinal injuries and underwent OC fusion on an emergency basis. Patients consisted of 10 males and 2 females with an average age of 47 years (range, 18–82 years). All patients sustained high-energy injuries. Three patients out of 6 patients with normal BAI (basion-axial interval) and BDI (basion-dens interval) values showed visible CCJ injuries on CT scans. However, the remaining 3 patients had no clear evidence of occipitoatlantal instability on CT scans. MRI clearly described several findings indicating occipitoatlantal instability. The 8 patients with normal values of ADI (atlanto-dens interval interval) demonstrated atlantoaxial instability on CT scan, however, all MRI more clearly and reliably demonstrated C1/2 facet injury and/or cruciate ligament injury. Conclusion: We advocate measures to help recognize CCJ injury at an early stage in the present study. Occipitoatlantal instability needs to be carefully investigated on MRI in addition to CT scan with special attention to facet joint and ligament integrity.

DOI： 10.14245/ns.2142860.430

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Scopus

PubMed

Language：English  

DOI： 10.21037/jss-21-83

PubMed

Language：Japanese   Publisher：Frontiers in Human Neuroscience  

Purpose: Maintenance of cognitive performance is important for healthy aging. This study aims to elucidate the relationship between brain networks and cognitive function in subjects maintaining relatively good cognitive performance. Methods: A total of 120 subjects, with equal number of participants from each age group between 20 and 70 years, were included in this study. Only participants with Addenbrooke’s Cognitive Examination – Revised (ACE-R) total score greater than 83 were included. Anatomical T1-weighted MR images and resting-state functional MR images (rsfMRIs) were taken from all participants using a 3-tesla MRI scanner. After preprocessing, several factors associated with age including the ACE-R total score, scores of five domains, sub-scores of ACE-R, and brain volumes were tested. Morphometric changes associated with age were analyzed using voxel based morphometry (VBM) and changes in resting state networks (RSNs) were examined using dual regression analysis. Results: Significant negative correlations with age were seen in the total gray matter volume (GMV, r = −0.58), and in the memory, attention, and visuospatial domains. Among the different sub-scores, the score of the delayed recall (DR) showed the highest negative correlation with age (r = −0.55, p < 0.001). In VBM analysis, widespread regions demonstrated negative correlation with age, but none with any of the cognitive scores. Quadratic approximations of cognitive scores as functions of age showed relatively delayed decline compared to total GMV loss. In dual regression analysis, some cognitive networks, including the dorsal default mode network, the lateral dorsal attention network, the right / left executive control network, the posterior salience network, and the language network, did not demonstrate negative correlation with age. Some regions in the sensorimotor networks showed positive correlation with the DR, memory, and fluency scores. Conclusion: Some domains of the cognitive test did not correlate with age, and even the highly correlated sub-scores such as the DR score, showed delayed decline compared to the loss of total GMV. Some RSNs, especially involving cognitive control regions, were relatively maintained with age. Furthermore, the scores of memory, fluency, and the DR were correlated with the within-network functional connectivity values of the sensorimotor network, which supported the importance of exercise for maintenance of cognition.

DOI： 10.3389/fnhum.2021.753836

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Scopus

PubMed

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

Aphasic status epilepticus (ASE) is a subtype of focal nonconvulsive status epilepticus, in which language disturbance is the only objective clinical manifestation. We present two cases of patients who experienced delayed onset of temporal aphasia after the removal of glioma at the language-dominant hemisphere. In both cases, arterial spin labeling was useful for diagnosis and antiepileptic drug was effective. ASE should be considered a cause of persistent aphasia after glioma resection at or near the language area.

DOI： 10.1007/s00701-021-04984-z

PubMed

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

BACKGROUND: Recent comprehensive studies have revealed several molecular alterations that are frequently found in meningiomas. However, effective treatment reagents targeting specific molecular alterations have not yet been identified because of the limited number of representative research models of meningiomas. METHODS: We performed organoid cultures using meningioma cells and meningioma tumor tissues. Using immunohistochemistry and molecular analyses consisting of whole-exome sequencing, RNA-seq, and DNA methylation analyses, we compared the histological findings and molecular profiling of organoid models with those of parental tumors. Further, using these organoid models together with a public database of meningiomas, we explored molecular alterations, which are a potent treatment target for meningioma. RESULTS: We established 18 organoid models comprising of two malignant meningioma cells (HKBMM and IOMM-Lee), 10 benign meningiomas, four malignant meningiomas, and two solitary fibrous tumors (SFTs). The organoids exhibited consistent histological features and molecular profiles with those of the parental tumors. Using a public database, we identified that upregulated forkhead box M1 (FOXM1) was correlated with increased tumor proliferation. Overexpression of FOXM1 in benign meningioma organoids increased organoid proliferation; depletion of FOXM1 in malignant organoids decreased proliferation. Additionally, thiostrepton, a FOXM1 inhibitor combined with radiation therapy, significantly inhibited the proliferation of malignant meningioma organoid models. CONCLUSIONS: An organoid model for meningioma enabled us to elucidate the tumor biology of meningioma along with potent treatment targets for meningioma.

DOI： 10.1093/neuonc/noab155

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Scopus

PubMed

Language：Japanese   Publisher：The Japan Neurosurgical Society  

<p>Magnetic resonance (MR)-guided focused ultrasound surgery (MRgFUS) is the latest minimally invasive stereotactic procedure, and thalamotomy using this novel modality has demonstrated its effectiveness and safety, especially for patients with essential tremor (ET) and Parkinson’s disease (PD). In Japan, the application of MRgFUS to treat ET and PD has recently been covered by health insurance. Technically, the transducer with 1024 elements emits ultrasound beams, which are then focused on the target with a phase control, resulting in optimal ablation by thermal coagulation. The technical advantages of MRgFUS are continuous intraoperative monitoring of clinical symptoms and MR images and fine adjustment of the target by the steering function. Postoperative tremor control is compatible with other modalities, although long-term follow-up is necessary. The adverse effects are usually transient and acceptable. Prognostic factors for good tremor control include high temperature and large lesion size. A high skull density ratio is a factor to achieve high temperature and large lesioning, but it may not be necessary and sufficient for clinical outcomes. For patients with advanced symptoms such as bilateral tremor or head/neck tremor, deep brain stimulation may be recommended because of the adjustability of stimulation and the possibility of bilateral treatment. Patients have high expectations of MRgFUS because of its non-invasiveness. To perform this treatment safely and effectively, physicians need to understand the technological aspects, the physiological principles. To choose the appropriate modality, physicians also should recognize the clinical advantages and disadvantages of MRgFUS compared to other modalities.</p>

DOI： 10.2176/nmc.ra.2021-0187

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Scopus

PubMed

Language：Japanese   Publisher：Biochemical and Biophysical Research Communications  

Oxytocin is known as a social bonding hormone, but it also functions as an anxiolytic or analgesic neurotransmitter. When oxytocin regulates pain or anxiousness centrally as a neurotransmitter, it is secreted by neurons and directly projected to targeted regions. Although the function of oxytocin at the spinal level is well studied, its effects at the supraspinal level are poorly understood. We aimed to investigate the effect of oxytocin at the supraspinal level in vivo using C57BL/6J (wild-type [WT]), oxytocin-deficient (Oxt−/−), oxytocin receptor-deficient (Oxtr−/−), and oxytocin receptor-Venus (OxtrVenus/+) mice lines. Response thresholds in Oxtr−/− mice in Hargreaves and von-Frey tests were significantly lower than those in WT mice, whereas open field and light/dark tests showed no significant differences. Moreover, response thresholds in Oxt−/− mice were raised to those in WT mice after oxytocin administration. Following the Hargreaves test, we observed the co-localisation of c-fos with Venus or the oxytocin receptor in the periaqueductal gray (PAG), medial amygdala (MeA), and nucleus accumbens (NAc) regions in OxtrVenus/+ mice. Furthermore, in the PAG, MeA, and NAc regions, the co-localisation of oxytocin with c-fos and gamma-aminobutyric acid was much stronger in Oxtr−/− mice than in WT mice. However, following von-Frey test, the same findings were observed only in the MeA and NAc regions. Our results suggest that oxytocin exerts its analgesic effect on painful stimulation via the PAG region and a self-protective effect on unpleasant stimulation via the MeA and NAc regions.

DOI： 10.1016/j.bbrc.2021.08.042

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Scopus

PubMed

Language：Japanese   Publisher：The Japan Neurosurgical Society  

<p>The carotid sinus reflex (CSR) is a rare complication of the Pipeline Embolization Device (PED) deployment. No study has assessed the potential risk factors in a case series. The purpose of this study was to examine CSR triggering during PED deployment. Thirty-seven consecutive patients who underwent PED deployment were included. All procedures were performed under local anesthesia with mild sedation. We retrospectively analyzed patient characteristics, PED deployment time, and vital signs during the procedure. The vital signs included the pulse rate (PR) and systolic blood pressure (SBP) obtained at three timepoints (pre-deployment, during deployment, post-deployment). We examined the triggering of the CSR during PED deployment by comparing the vital signs at the three timepoints. Moreover, risk factors for CSR were analyzed with univariate analysis. The patients’ average age was 66.3 years. The average size of the aneurysm was 18.0 mm. Six patients (16.2%) showed a decline in the SBP or PR defined as CSR. One patient had a transient cardiac arrest and two had severe transient bradycardia. Deployment into the ophthalmic segment of the internal carotid artery (C2 segment) aneurysm (p = 0.022), prolonged PED deployment time more than 14.5 minutes (p = 0.005), and an acute angle of the anterior genu less than 51.5 degrees (p = 0.005) were risk factors in triggering CSR. CSR may be triggered during PED deployment under local anesthesia with mild sedation. Deployment to the C2 segment aneurysm, prolonged PED deployment time, and an acute angle of the anterior genu were associated with CSR triggering.</p>

DOI： 10.2176/nmc.oa.2021-0049

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Scopus

PubMed

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

DOI： 10.1507/endocrine.97.s.hpt_49

CiNii Research

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

DOI： 10.1507/endocrine.97.s.hpt_16

CiNii Research

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

Isocitrate dehydrogenase-mutant low-grade gliomas (IDHmut-LGG) grow slowly but frequently undergo malignant transformation, which eventually leads to premature death. Chemotherapy and radiotherapy treatments prolong survival, but can also induce genetic (or epigenetic) alterations involved in transformation. Here, we developed a mathematical model of tumor progression based on serial tumor volume data and treatment history of 276 IDHmut-LGGs classified by chromosome 1p/19q codeletion (IDHmut/1p19qcodel and IDHmut/1p19qnoncodel) and performed genome-wide mutational analyses, including targeted sequencing and longitudinal whole-exome sequencing data. These analyses showed that tumor mutational burden correlated positively with malignant transformation rate, and chemotherapy and radiotherapy significantly suppressed tumor growth but increased malignant transformation rate per cell by 1.8 to 2.8 times compared with before treatment. This model revealed that prompt adjuvant chemoradiotherapy prolonged malignant transformation-free survival in small IDHmut-LGGs (≤ 50 cm3). Furthermore, optimal treatment differed according to genetic alterations for large IDHmut-LGGs (> 50 cm3); adjuvant therapies delayed malignant transformation in IDHmut/1p19qnoncodel but often accelerated it in IDHmut/1p19qcodel. Notably, PI3K mutation was not associated with malignant transformation but increased net postoperative proliferation rate and decreased malignant transformation-free survival, prompting the need for adjuvant therapy in IDHmut/1p19qcodel. Overall, this model uncovered therapeutic strategies that could prevent malignant transformation and, consequently, improve overall survival in patients with IDHmut-LGGs. SIGNIFICANCE: A mathematical model successfully estimates malignant transformation-free survival and reveals a link between genetic alterations and progression, identifying precision medicine approaches for optimal treatment of IDH-mutant low-grade gliomas.

DOI： 10.1158/0008-5472.CAN-21-0985

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Scopus

PubMed

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

In cases of malignant gliomas located at language eloquent area, it is often difficult to preoperatively detect those area with functional MRI. Awake surgery is often used to spare the language eloquent area during surgery for such tumors; it is not available for a patient whose intracranial pressure is elevated due to the malignant tumor. The Wada test involves infusing anesthetic agents into the internal carotid artery to determine language dominancy before surgery for epilepsy or brain tumor. The super-selective Wada test is a technique to detect more detailed functional localization by infusing anesthetics into far distal middle cerebral artery branches. We present a 37-year-old man suffering from a left frontal lobe glioblastoma, in whom detection of an artery supplying Broca's area was attempted by a super-selective Wada test. The super-selective Wada test successfully detected the branch of middle cerebral artery supplying Broca's area. Total resection of the contrast-enhancing area was achieved without damaging the artery supplying Broca's area without any neurological sequelae. This is the first report describing the usefulness of the super-selective Wada test in glioblastoma treatment. Our findings suggest that the super-selective Wada test is a powerful and useful means to distinguish the artery that supplies the language area from the tumor feeding artery in cases of tumors in the language eloquent area.

DOI： 10.2176/nmc.tn.2021-0054

PubMed

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

DOI： 10.1007/s10014-021-00406-1

PubMed

Other Link： https://link.springer.com/article/10.1007/s10014-021-00406-1/fulltext.html

Language：Japanese   Publisher：株式会社医学書院  

DOI： 10.11477/mf.1436204463

PubMed

CiNii Research

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

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

PURPOSE: The aim of this study was to assess the effect of the extent of resection (EOR) of tumors on survival in a series of patients with grade II and III gliomas (GII/III-gliomas) who underwent awake brain mapping. METHODS: We retrospectively analyzed 126 patients with GII/III-gliomas in the dominant and non-dominant hemisphere who underwent awake brain surgery at the same institution between December 2012 and May 2020. RESULTS: EOR cut-off values for improved progression-free survival (PFS) were determined by a receiver operator characteristic (ROC) analysis of 5-year PFS. The ROC for EOR showed a cut-off value of ≥ 85.3%. The median PFS rate of patients with GII/III-gliomas in the group with an EOR ≥ 100%, including supratotal resection (n = 47; median survival [MS], not reached), was significantly higher than that in the group with an EOR < 90% (n = 52; MS, 43.1 months; 95% CI 37.7-48.5 months; p = 0.03). In patients with diffuse astrocytomas and anaplastic astrocytomas, the group with EOR ≥ 100%, including supratotal resection (n = 25; MS, not reached), demonstrated a significantly better PFS rate than did the group with an EOR < 100% (n = 45; MS, 35.8 months; 95% CI 19.9-51.6 months; p = 0.03). Supratotal or gross total resection was correlated with better PFS in IDH-mutant type of diffuse astrocytomas and anaplastic astrocytomas (n = 19; MS, not reached vs. n = 35; MS, 40.6 months; 95% CI 22.3-59.0 months; p = 0.02). By contrast, supratotal or gross total resection was not associated with longer PFS rates in patients with IDH-wild type of diffuse astrocytomas and anaplastic astrocytomas. CONCLUSIONS: The present study demonstrates a significant association between tumor EOR and survival in patients with GII/III gliomas. The EOR cut-off value for 5-year PFS was ≥ 85.3%. It is noteworthy that supratotal or gross total resection significantly correlated with better PFS in IDH-mutant type of WHO grade II and III astrocytic tumors. In light of our finding that EOR did not correlate with PFS in patients with aggressive IDH-wild type of diffuse astrocytomas and anaplastic astrocytomas, we suggest treatments that are more intensive will be needed for the control of these tumors.

DOI： 10.1007/s11060-021-03776-w

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Scopus

PubMed

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

DOI： 10.1007/s00701-021-04737-y

PubMed

Other Link： https://link.springer.com/article/10.1007/s00701-021-04737-y/fulltext.html

Language：Japanese   Publisher：株式会社医学書院  

DOI： 10.11477/mf.1436204432

PubMed

CiNii Research

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

OBJECTIVE: We developed an actuator-driven pulsed water jet device (ADPJ) for flexible neuroendoscopy to achieve effective tissue dissection with vasculature preservation. Although flexibility is a strong advantage for minimally invasiveness, the effect of the ductile curvature on the dissection profiles remains unknown. The purpose of this study was to clarify the impact of the curvature change of the ADPJ connecting tube on the dissection safety and efficacy. RESULTS: Three ADPJ connecting tubes with different inner diameters (1.0, 0.75, 0.5 mm) were used to dissect the brain phantom. They were bent at 3 angles: 0°, 60°, and 120°. The dissection profiles were evaluated using the mean depth and coefficient of variation (CV) for efficacy and safety, respectively.The larger inner diameter connecting tube dissected more deeply. The dissection depth was not changed regardless of the curvature degree in each tube. There was no significant difference in CVs regardless of inner diameter and curvature. The ductile curvature of the flexible neuroendoscope did not affect the efficacy and safety of the ADPJ dissection profile. Among the numerous instruments, tube-formed devices, including suction and injecting devices such as ADPJ, can be used safely and effectively without flexibility-related limitations.

DOI： 10.1186/s13104-021-05475-1

PubMed

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

INTRODUCTION: The purpose of this study is to clarify the clinical features of temozolomide (TMZ)-related hepatitis B virus (HBV) reactivation and to identify HBV reactivation predictive factors. METHOD: We retrospectively reviewed the clinical course of 145 patients newly diagnosed or with recurrent malignant glioma treated with TMZ. Before treatment, we screened patients for HB surface antigen (HBsAg) positivity (HBV carrier) and HBsAg negativity. Patients were also screened for antibody for HB core antigen (anti-HBc) positivity and/or for HB surface antigen positivity (resolved HBV infection). The patients were monitored by HBV DNA, alanine, and aspartate aminotransaminase during and after the completion of TMZ. HBV carriers and those with resolved HBV infections with HBV reactivation received preemptive entecavir treatment. In those with resolved HBV infections, we analyzed clinical characters for the predictive factors for HBV reactivation. RESULTS: In one of two HBV carriers, HBV DNA turned positive 8 months after the completion of TMZ and entecavir. In four (16.7%) of 24 resolved HBV infections, HBV DNA turned detectable at completion of concomitant radiation and TMZ or during monthly TMZ. HBV DNA turned negative with entecavir in all patients without liver dysfunction. In resolved HBV infections, those with a high anti-HBc titer had significantly higher incidence of HBV reactivation than those with low anti-HBc titers (60% vs. 5.3%: p = 0.018). CONCLUSION: Screenings, monitoring, and preemptive entecavir were important for preventing TMZ-related HBV reactivations. Anti-HBc titers could be the predictive markers for HBV reactivation in the those with resolved HBV infections.

DOI： 10.1007/s10147-020-01814-7

PubMed

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

<title>Abstract</title>
<sec>
<title>Background</title>
Thalamic high-grade gliomas (HGGs) are rare tumors with a dismal prognosis. H3K27M and telomerase reverse transcriptase promoter (TERTp) mutations reportedly contribute to poor prognoses in HGG cases. We investigated the outcomes of surgically treated adult thalamic HGGs to evaluate the prognostic significance of H3K27M and TERTp mutations.


</sec>
<sec>
<title>Methods</title>
We retrospectively analyzed 25 adult patients with thalamic HGG who underwent maximum surgical resection from January 1997 to March 2020. The histological and molecular characteristics, progression-free survival (PFS), and overall survival (OS) of the patients were compared. For molecular characteristics, target sequencing was used to determine the H3F3A, HIST1H3B, and TERTp mutations.


</sec>
<sec>
<title>Results</title>
H3K27M mutations were detected in 12/25 (48.0%) patients. TERTp mutations were not detected in H3K27M-mutant gliomas but were detected in 8/13 (61.5%) of H3 wild-type gliomas. Although it was not significant, H3K27M-mutant gliomas tended to have a shorter PFS (6.7 vs 13.1 months; P = .2928) and OS (22.8 vs 24.4 months; P = .2875) than H3 wild-type gliomas. Moreover, the prognosis of TERTp-mutant gliomas was as poor as that of H3K27M-mutant gliomas. Contrary, 5 gliomas harboring both H3 and TERTp wild-type showed a better median PFS (59.2 vs 6.4 months; P = .0456) and OS (71.8 vs 24.4 months; P = .1168) than those harboring H3K27M or TERTp mutations.


</sec>
<sec>
<title>Conclusions</title>
TERTp-mutant gliomas included in the H3 wild-type glioma group limited patient survival as they exhibited an aggressive course similar to H3K27M-mutant gliomas. Comprehensive molecular work-up for the H3 wild-type cases may further confirm this finding.


</sec>

DOI： 10.1093/noajnl/vdab038

PubMed

Other Link： http://academic.oup.com/noa/article-pdf/3/1/vdab038/37929606/vdab038.pdf

Language：English  

DOI： 10.2176/nmccrj.cr.2020-0369

PubMed

Language：Japanese   Publisher：The Japanese Society for Neuroendovascular Therapy  

<p><b>Objective</b>: Herein, we report a case of multiple dural arteriovenous fistulas (DAVFs) in the left superior petrosal sinus (SPS) successfully treated by transvenous embolization via the right SPS.</p><p><b>Case Presentation</b>: A 60-year-old woman was diagnosed with multiple DAVFs with deep venous reflux to evaluate consciousness disturbance. We performed packing of the left transverse sinus, which was chiefly involved in reflux to the straight sinus. However, consciousness continued to be mildly disturbed, and DSA was performed again. A shunt was inserted into the left SPS, and the subsequently occurring deep venous reflux was clearly revealed. Because the ipsilateral transverse sinus and both inferior petrosal sinuses were already occluded and unavailable as access routes, we used the contralateral SPS and both cavernous sinuses to navigate the microcatheter into the shunt. We achieved occlusion of the SPS and stopped the reflux by inserting the intermediate catheter firmly into the bilateral SPS.</p><p><b>Conclusion</b>: An access route through the contralateral SPS might successfully enable transvenous embolization of DAVF cases without an ipsilateral access route.</p>

DOI： 10.20626/nkc.tn.2021-0009

CiNii Research

Language：Japanese   Publisher：日本神経治療学会  

DOI： 10.15082/jsnt.38.6_s211

CiNii Research

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

TERT promoter mutations are commonly associated with 1p/19q codeletion in IDH-mutated gliomas. However, whether these mutations have an impact on patient survival independent of 1p/19q codeletion is unknown. In this study, we investigated the impact of TERT promoter mutations on survival in IDH-mutated glioma cases. Detailed clinical information and molecular status data were collected for a cohort of 560 adult patients with IDH-mutated gliomas. Among these patients, 279 had both TERT promoter mutation and 1p/19q codeletion, while 30 had either TERT promoter mutation (n = 24) or 1p/19q codeletion (n = 6) alone. A univariable Cox proportional hazard analysis for survival using clinical and genetic factors indicated that a Karnofsky performance status score (KPS) of 90 or 100, WHO grade II or III, TERT promoter mutation, 1p/19q codeletion, radiation therapy, and extent of resection (90–100%) were associated with favorable prognosis (p < 0.05). A multivariable Cox regression model revealed that TERT promoter mutation had a significantly favorable prognostic impact (hazard ratio = 0.421, p = 0.049), while 1p/19q codeletion did not have a significant impact (hazard ratio = 0.648, p = 0.349). Analyses incorporating patient clinical and genetic information were further conducted to identify subgroups showing the favorable prognostic impact of TERT promoter mutation. Among the grade II-III glioma patients with a KPS score of 90 or 100, those with IDH-TERT co-mutation and intact 1p/19q (n = 17) showed significantly longer survival than those with IDH mutation, wild-type TERT, and intact 1p/19q (n = 185) (5-year overall survival, 94% and 77%, respectively; p = 0.032). Our results demonstrate that TERT promoter mutation predicts favorable prognosis independent of 1p/19q codeletion in IDH-mutated gliomas. Combined with its adverse effect on survival among IDH-wild glioma cases, the bivalent prognostic impact of TERT promoter mutation may help further refine the molecular diagnosis and prognostication of diffuse gliomas.

DOI： 10.1186/s40478-020-01078-2

Scopus

PubMed

Publishing type：Research paper (scientific journal)  

Multinodular and vacuolating neuronal tumor (MVNT) is a recently recognized type of neural tumor that was first documented in 2013. This tumor type is characterized by multiple nodules of neuronal cells with vacuolar degeneration localized in the subcortical white matter. Here, we report a case of MVNT in the right temporal lobe of a 26-year-old female. Magnetic resonance imaging (MRI) revealed a high-intensity lesion on a T2- weighted image of the right temporal lobe without contrast enhancement. Positron emission tomography revealed increased 11C-methionine uptake in the lesion, which was pathologically diagnosed as MVNT.

DOI： 10.1016/j.inat.2020.100861

Scopus

Language：English   Publisher：(一社)日本小児血液・がん学会  

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

INTRODUCTION: Anaplastic astrocytoma has a dismal prognosis with conventional treatment. Multidisciplinary treatment is needed to control the disease; however, side effects of the treatment reduce a patient's quality of life (QOL). Carmustine-impregnated wafers (Gliadel®, Eisai Co., Ltd., Tokyo, Japan), one of the treatment modalities for anaplastic astrocytoma, has been reported to have drug-induced fever as a side effect. CASE REPORT: A 36-year-old man underwent excision for a recurrent brain tumor. Histopathological examination established a diagnosis of anaplastic astrocytoma and an intracranial carmustine implant was placed for local chemotherapy. Postoperatively, the patient developed high fever, which could not be controlled using antipyretics. The high fever ameliorated dramatically after the administration of Kampo medicines, specifically orengedokuto and shosaikoto, and the patient could continue chemotherapy. CONCLUSION: To the best of our knowledge, this is the first report of successful treatment of intractable carmustine implant-induced fever using Kampo medicine. In this case, Kampo medicine led to an improvement of QOL.

DOI： 10.1016/j.explore.2020.08.014

PubMed

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

Most cases of optic hypothalamic pilocytic astrocytoma (OHPA) develop during childhood, so few cases of histologically verified OHPA have been described in adolescents and young adults (AYA). To elucidate the clinical features of OHPA with histological verification in AYA, we reviewed the clinical and radiological finding of OHPA treated at our institute from January 1997 and July 2017. AYA are aged between 15 and 39 years. The clinical courses of 11 AYA patients with optic hypothalamic glioma (OHG) without neurofibromatosis type 1 were retrospectively reviewed. About six patients were diagnosed in childhood and followed up after 15 years of age, and five patients developed OHPA during AYA. Histological diagnosis, verified at initial presentation or recurrence, was pilocytic astrocytoma in 10 and pilomyxoid astrocytoma in one. After initial treatment including debulking surgery and/or chemotherapy, tumor progression occurred 16 times in seven patients as cyst formation, tumor growth, and intratumoral hemorrhage. Five of 10 patients suffered deterioration of visual function during AYA. One of 10 cases had endocrinopathies requiring hormone replacement at last follow-up examination. In conclusion, histological diagnoses of OHG before and in AYA were pilocytic astrocytoma or pilomyxoid astrocytoma. Both pediatric and AYA-onset OHPA demonstrate high incidences of tumor progression and visual dysfunctions in AYA, so that long-term follow up is essential after the completion of treatment for pediatric and AYA-onset OHPA. The optimal timing of debulking surgery and radiation therapy should be established to achieve the long-term tumor control and to preserve the visual function.

DOI： 10.2176/nmc.oa.2019-0208

PubMed

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

DOI： 10.1093/ons/opz172

PubMed

Language：Japanese   Publisher：(公社)日本小児科学会  

Language：Japanese   Publisher：(公社)日本小児科学会  

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

<title>Abstract</title>
<sec>
<title>Background</title>
Treatment options for patients suffering brainstem gliomas are quite limited as surgery is not an option against intrinsic tumors at brainstem and chemotherapy generally failed to demonstrate its efficacy. Intracerebral convection-enhanced delivery (CED) is a novel approach for administering chemotherapy to patients with brain tumors. We present the results of phase I trial of CED of nimustine hydrochloride (ACNU), designed to determine the maximum tolerable concentration of ACNU, for patients with recurrent brainstem gliomas.


</sec>
<sec>
<title>Methods</title>
Sixteen patients, aged 3–81 years old, suffering from recurrent brainstem gliomas, including diffuse intrinsic pontine glioma patients as well as patients with recurrent gliomas that originated from non-brainstem sites, were enrolled in this trial between February 2011 and April 2016. The dose/concentration escalation trial included 3 dose/concentration groups (0.25, 0.5, and 0.75 mg/mL, all at 7 mL) to determine the safety and tolerability of CED of ACNU. Real-time monitoring of drug distribution was performed by mixing gadolinium-tetraazacyclododecanetetraacetic acid (Gd-DOTA) in the infusion solution. CED of ACNU was given in combination with oral or intravenous temozolomide chemotherapy.


</sec>
<sec>
<title>Results</title>
CED of ACNU demonstrated antitumor activity, as assessed by radiographic changes and prolonged overall survival. The recommended dosage was 0.75 mg/mL. Drug-associated toxicity was minimal.


</sec>
<sec>
<title>Conclusions</title>
Intracerebral CED of ACNU under real-time monitoring of drug distribution, in combination with systemic temozolomide, was well tolerated among patients with recurrent brainstem gliomas. The safety and efficacy observed suggest the clinical benefits of this strategy against this devastating disease. Based on this phase I study, further clinical development of ACNU is warranted.


</sec>

DOI： 10.1093/noajnl/vdaa033

PubMed

Other Link： http://academic.oup.com/noa/article-pdf/2/1/vdaa033/33171642/vdaa033.pdf

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

<title>Abstract</title>
<sec>
<title>Background</title>
Although mutations in the promoter region of the telomerase reverse transcriptase (TERTp) gene are the most common alterations in glioblastoma (GBM), their clinical significance remains unclear. Therefore, we investigated the impact of TERTp status on patient outcome and clinicopathological features in patients with GBM over a long period of follow-up.


</sec>
<sec>
<title>Methods</title>
We retrospectively analyzed 153 cases of GBM. Six patients with isocitrate dehydrogenase 1 (IDH1) or H3F3A gene mutations were excluded from this study. Among the 147 cases of IDH wild-type GBM, 92 (62.6%) had the TERTp mutation. Clinical, immunohistochemical, and genetic factors (BRAF, TP53 gene mutation, CD133, ATRX expression, O6-methylguanine-DNA methyltransferase [MGMT] promoter methylation) and copy number alterations (CNAs) were investigated.


</sec>
<sec>
<title>Results</title>
GBM patients with the TERTp mutation were older at first diagnosis versus those with TERTp wild type (66.0 vs. 60.0 years, respectively, P = .034), and had shorter progression-free survival (7 vs. 10 months, respectively, P = .015) and overall survival (16 vs. 24 months, respectively, P = .017). Notably, magnetic resonance imaging performed showed that TERTp-mutant GBM was strongly associated with multifocal/distant lesions (P = .004). According to the CNA analysis, TERTp mutations were positively correlated with EGFR amp/gain, CDKN2A deletion, and PTEN deletion; however, these mutations were negatively correlated with PDGFR amp/gain, CDK4 gain, and TP53 deletion.


</sec>
<sec>
<title>Conclusions</title>
TERTp mutations were strongly correlated with multifocal/distant lesions and poor prognosis in patients with IDH wild-type GBM. Less aggressive GBM with TERTp wild type may be a distinct clinical and molecular subtype of IDH wild-type GBM.


</sec>

DOI： 10.1093/noajnl/vdaa114

PubMed

Other Link： http://academic.oup.com/noa/article-pdf/2/1/vdaa114/35106481/vdaa114.pdf

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

DOI： 10.2176/nmccrj.cr.2019-0043

PubMed

Language：English   Publishing type：Research paper (scientific journal)   Publisher：OXFORD UNIV PRESS  

A randomized phase III trial in Japan commenced in June 2019. The present standard treatment for newly diagnosed glioblastoma is maximal resection followed by chemoradiotherapy with temozolomide. The purpose of this study is to confirm the superiority of maximal resection with carmustine wafer implantation followed by chemoradiotherapy with temozolomide over the standard maximal resection followed by chemoradiotherapy with temozolomide in terms of overall survival for newly diagnosed glioblastoma. A total of 250 patients will be accrued from 35 Japanese institutions in 5.5 years. Patients with >90% surgical resection will be registered and randomly assigned to each group with 1:1 allocation. The primary endpoint is overall survival and the secondary endpoints are progression-free survival, loco-regional progression-free survival and incidence of adverse events. This trial has been registered in the Japan Registry of Clinical Trial, as jRCT1031190035 [https://jrct.niph.go.jp/en-latest-detail/jRCT1031190035].

DOI： 10.1093/jjco/hyz169

Web of Science

PubMed

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

After introduction of levetiracetam (LEV), treatment of seizures in patients with malignant brain tumors has prominently improved. On the other hand, we still experience some cases with LEV-uncontrollable epilepsy. Perampanel (PER) is a noncompetitive a-amino-3-hydroxy-5-methyl-4-isoaxazolepropionate acid receptor antagonist that has recently been approved for treating focal epilepsy as a secondary drug of choice. Available literature reporting PER medication in patients with gliomas is still sparse. Here, we report our initial experience with glioma patients and report efficacy of adding low dose 2–4 mg PER to LEV in patients whose seizure were uncontrollable with LEV monotherapy. Clinical outcome data of 18 consecutive patients were reviewed. This included nine males and nine females aged 24–76 years (median, 48.5 years), treated for glioma between June 2009 to December 2018. We added PER to patients with LEV-uncontrollable epilepsy. Adverse effects, irritability occurred in two patients, but continuous administration was possible in all cases. Though epileptic seizures occurred in four cases receiving 2 mg PER, 17 cases achieved seizure freedom by dose increments; final dose, 2–4 mg PER added to LEV 500–3000 mg. Our study revealed anti-epileptic efficacy of low dose PER 2–4 mg as first add-on therapy to LEV in glioma patients who have failed or intolerable to LEV monotherapy. Low dose PER added on to LEV may have favorable efficacy with tolerable adverse effects in glioma patients with LEV-uncontrollable epilepsy.

DOI： 10.2176/nmc.oa.2018-0245

Scopus

PubMed

Language：Japanese   Publisher：(NPO)日本脳神経血管内治療学会  

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

Background: Chronic subdural hematoma (CSDH) is a common neurosurgical disease. A subset of patients with CSDH may exhibit underlying spontaneous intracranial hypotension (SIH). Bilateral CSDH has a causal relationship with SIH, but there is no known causal relationship between unilateral CSDH and SIH.Case description: We encountered four cases of unilateral CSDH due to SIH. The patients' age ranged between 44 and 64 years; there were three males and one female. All patients presented with headache as their initial symptom, and then became comatose. Computed tomography demonstrated unilateral CSDH and transtentorial herniation in all patients. Treatments were emergency epidural blood patch (EBP) and evacuation of CSDH. The site of cerebrospinal fluid leak could not be identified in three patients; therefore, EBP was performed at upper and lower spine. All patients recovered from SIH; however, one patient experienced poor outcome due to Duret hemorrhage and ischemic complications of transtentorial herniation. Cranial asymmetry was present in all four patients, and unilateral CSDH was located on the side of the most curved cranial convexity.Conclusions: Unilateral CSDH, asymmetric cranial morphology, and transtentorial herniation in relatively young patients may indicate underlying SIH.

DOI： 10.1080/02688697.2019.1667482

PubMed

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

DOI： 10.1016/j.wneu.2019.04.015

PubMed

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

Web of Science

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

Language：English  

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

PURPOSE: Intracranial glioblastomas with simultaneous spinal lesions prior to chemoradiation therapy or craniotomy, defined as initial spinal metastasis, are not well understood. Herein, we investigated intracranial glioblastoma and demonstrated the importance of spinal screening using gadolinium enhanced spinal magnetic resonance imaging (Gd-MRI). METHODS: Consecutive adult patients with intracranial glioblastoma were treated between 2010 and 2014 and received spinal screening using Gd-MRI. Spinal screening was performed regardless of spine-related symptoms, and patients presenting with and without initial spinal metastasis (spinal and non-spinal groups, respectively) were compared based on patient demographics, tumor characteristics, radiological and molecular features, and overall survival (OS). RESULTS: During the study period, 116 glioblastoma cases were treated and 87 of these (76%) underwent spinal screening. Among these patients, 11 (13%) were included in the spinal group, and 76 (87%) were included in the non-spinal group. All patients of the spinal group were free of symptoms related to spinal lesions. Compared with the non-spinal group, intracranial lesions of the spinal group presented higher incidences of intracranial dissemination and were located at subventricular zones (P = 0.0012 and 0.020, respectively). MIB-1 labeling index, molecular alterations such as IDH1 mutation, TERT promoter mutation, and immunoreactivity of ATRX and MGMT did not differ between two groups. OS was significantly shorter in the spinal group than in the non-spinal group (P = 0.0054). CONCLUSIONS: This study revealed a relatively high incidence of spinal metastasis. A subset of glioblastoma patients benefited from spinal screening, through which early detection of asymptomatic spinal metastasis was achieved.

DOI： 10.1007/s11060-018-03036-4

PubMed

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

Extensive molecular analyses of ependymal tumors have revealed that supratentorial and posterior fossa ependymomas have distinct molecular profiles and are likely to be different diseases. The presence of C11orf95-RELA fusion genes in a subset of supratentorial ependymomas (ST-EPN) indicated the existence of molecular subgroups. However, the pathogenesis of RELA fusion-negative ependymomas remains elusive. To investigate the molecular pathogenesis of these tumors and validate the molecular classification of ependymal tumors, we conducted thorough molecular analyses of 113 locally diagnosed ependymal tumors from 107 patients in the Japan Pediatric Molecular Neuro-Oncology Group. All tumors were histopathologically reviewed and 12 tumors were re-classified as non-ependymomas. A combination of RT-PCR, FISH, and RNA sequencing identified RELA fusion in 19 of 29 histologically verified ST-EPN cases, whereas another case was diagnosed as ependymoma RELA fusion-positive via the methylation classifier (68.9%). Among the 9 RELA fusion-negative ST-EPN cases, either the YAP1 fusion, BCOR tandem duplication, EP300-BCORL1 fusion, or FOXO1-STK24 fusion was detected in single cases. Methylation classification did not identify a consistent molecular class within this group. Genome-wide methylation profiling successfully sub-classified posterior fossa ependymoma (PF-EPN) into PF-EPN-A (PFA) and PF-EPN-B (PFB). A multivariate analysis using Cox regression confirmed that PFA was the sole molecular marker which was independently associated with patient survival. A clinically applicable pyrosequencing assay was developed to determine the PFB subgroup with 100% specificity using the methylation status of 3 genes, CRIP1, DRD4 and LBX2. Our results emphasized the significance of molecular classification in the diagnosis of ependymomas. RELA fusion-negative ST-EPN appear to be a heterogeneous group of tumors that do not fall into any of the existing molecular subgroups and are unlikely to form a single category.

DOI： 10.1186/s40478-018-0630-1

PubMed

Publishing type：Research paper (international conference proceedings)  

We developed a new ultrasound-facilitated delivery (UFD) device with a drug infusion sapphire needle enabling real-time magnetic resonance imaging (MRI) observation and an ultrasound piezoelectric transducer (drive frequency: 260 kHz) with an effective structure for increasing drug diffusion. The sapphire needle (outside diameter: 0.6 mm, inside diameter: 0.3 mm, length: 150 mm) was grown by a micro-pulling-down method. The transducer is composed of two-stacked layers of lead zirconate titanate (PZT) on a brass base. The sound pressure distribution of the developed UFD device in water was measured with a miniature probe hydrophone, showing good agreement with the value simulated by the finite element method (FEM) analysis. The developed UFD device demonstrated high efficiency and reliability of drug diffusion, exhibiting an 8.5% increase in the volume of drug diffusion with 80% higher reproducibility and 66% lower driving voltage than the previous device. Consequently, the operating temperature of the new UFD device was decreased from 60 °C to room temperature.

DOI： 10.7567/JJAP.57.11UD07

Web of Science

Scopus

Web of Science

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

This report presents a case of stroke-like migraine attacks after radiation therapy (SMART) syndrome in a 31-year-old man in whom symptoms and radiological findings resolved with steroid pulsed therapy and reviews the literatures with special emphasis on the use of steroids against SMART syndrome. The patient had a past history of left temporal anaplastic astrocytoma and was treated with surgery followed by local 72 Gy radiation therapy and chemotherapy using Nimustine Hydrochloride. Four years after the surgery, he was suffering from subacute progressing symptoms of headache, right hemianopia, right hemiparesis and aphasia from 2 to 4 days before admission to our hospital. At first he was diagnosed as symptomatic epilepsy but after extensive examination, the final diagnosis was SMART syndrome. His symptoms soon improved with steroid pulse therapy. In the literature, steroid pulse therapy is not necessarily a standard of care for SMART syndrome, but it seemed to decrease the need of biopsy. As the lesions of SMART syndrome require differential diagnosis from recurrences, biopsy was performed in some cases. However, lack of benefit and possible detriment is reported with biopsy of SMART lesions. Through this experience we suggest that steroid pulse therapy may provide speedy recovery from symptoms, and it should be considered before other invasive investigations or treatments.

DOI： 10.1016/j.ensci.2018.05.003

PubMed

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

This study retrospectively reviewed our single institute experience to clarify the optimal indication and timing of salvage surgery. Retrospective analysis of 159 consecutive cases with germ cell tumors identified 20 cases with salvage surgery. These cases were classified based on the radiological response to neoadjuvant treatment before salvage surgery into increase (growing group, five cases), no change (stable group, seven cases), and decrease (shrinkage group, eight cases) in tumor size. Changes in tumor markers, histological findings, and the pattern of failure after salvage surgery were reviewed. Growing teratoma syndrome (GTS) is defined as enlargement of tumor consisting of mature teratoma after chemotherapy with normalization of tumor markers. In growing group, two cases presented GTS, whereas other three cases did not fulfill the criteria for GTS. All cases in stable and shrinkage group had elevated levels of tumor markers at presentation and decreased levels after neoadjuvant treatment. Histologically, sparse components of mature teratoma with extensive fibrosis were found in cases with GTS and seven of eight cases in shrinkage group, whereas mature teratoma without fibrosis was found in six of seven cases in stable group. Six cases recurred after salvage surgery. We identified three factors as risks for recurrence after salvage surgery, as follows: (1) growing lesion which did not fulfill the criteria for GTS, (2) non-normalized level of tumor marker before salvage surgery, and (3) residual germinoma component. In conclusion, salvage surgery is recommended for patients with GTS, or with normalized tumor markers in stable or shrinkage group.

DOI： 10.1007/s11060-018-2827-3

PubMed

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

INTRODUCTION: Ventriculoperitoneal(VP)shunts function because of the pressure differential between the intracranial space and the peritoneal cavity. Although chronic constipation is often a cause of VP shunt dysfunction in children, it is not well recognized in adults. We present a case of shunt dysfunction that not only resolved after resolution of constipation but also resulted in overdrainage. CASE REPORT: A 28-year-old woman who had a VP shunt placed 11 years prior for hydrocephalus was referred to our department because of an enlarged ventricle diagnosed with computed tomography(CT). She had a previous history of pineal germinoma and a VP shunt was placed to treat the associated hydrocephalus. At presentation, she complained of headache and somnolence. Shunt dysfunction was suspected, but no problem was detected in the shunt system, including pressure settings. As constipation was detected, we treated this condition. Soon after, her symptoms resolved and ventricle size normalized. She was discharged without any deficit, but overdrainage was detected with CT obtained 1 month later. CONCLUSION: Although constipation is not a common cause of shunt dysfunction in adult patients, it is important to consider to avoid unnecessary shunt revision. In the present case, resolution of chronic constipation resulted in resolution of shunt dysfunction. This suggests the importance of resolution of constipation in case of shunt adjustment, even in adult cases.

DOI： 10.11477/mf.1436203739

PubMed

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

OBJECTIVE: The purpose of this study was to clarify clinical features, outcomes, and the role of tumor resection in precocious, synchronous, and metachronous brain metastases. METHODS: Brain metastases were found before primary cancer detection in the precocious group, within 2 months after primary cancer detection in the synchronous group, and 2 months or later after primary cancer detection in the metachronous group. RESULTS: Of 471 patients with brain metastases, 93 (20%) were included in the precocious group, 76 (16%) in the synchronous group, and 302 (64%) in the metachronous group. The precocious group tended to be symptomatic, show a low Karnofsky Performance Status, and have a large single tumor, infrequent extracranial metastases, and frequent tumor resection compared with the other 2 groups. There were no differences in overall survival from the detection of brain metastases among the 3 groups in univariate and multivariate analyses. Of 471 cases, 97 (21%) underwent surgeries. Among this surgical cohort, overall survival from surgery was significantly shorter in the precocious group than in the metachronous group (P = 0.039). After adjustment for age, sex, tumor size, primary cancer, and the Graded Prognostic Assessment score, the hazard ratio for metachronous metastases was 0.52 (confidence interval, 0.29-0.95; P = 0.035). CONCLUSIONS: The timing of brain metastasis diagnosis is not a modifiable factor but affects patient demographics and treatment strategies. In particular, the precocious group is a unique subset of brain metastases that require special consideration during clinical decision making.

DOI： 10.1016/j.wneu.2017.10.145

PubMed

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

DOI： 10.11477/mf.1436203736

PubMed

Language：English   Publishing type：Research paper (scientific journal)   Publisher：Springer Tokyo  

Radiation therapy is sometimes performed to control intracranial acute lymphoblastic leukemia (ALL), but may lead to radiation-induced malignant glioma. The clinical, radiological, histological, and molecular findings are described of three cases of radiation-induced glioblastoma after the treatment for ALL. They received radiation therapy at age 6–8 years. The latency from radiation therapy to the onset of radiation-induced glioblastoma was 5–10 years. Magnetic resonance imaging demonstrated diffuse lesions with multiple small enhanced lesions in all cases. Histological examination showed that the tumors consisted of mainly small round astrocytic atypical cells in one case, and astrocytic atypical cells with elongated cytoplasm and nuclear pleomorphism with small cell component in two cases. Microvascular proliferation was present in all cases. Immunohistochemical analysis for B-Raf V600E, and mutational analysis for the isocitrate dehydrogenase (IDH) 1, IDH2, and H3F3A gene revealed the wild-type alleles in all three cases. The integrated diagnoses were IDH wild-type glioblastoma, and local irradiation and concomitant temozolomide were performed. After the initial treatment, significant shrinkage of the diffuse lesion and enhanced lesion was found in all cases. Radiation-induced glioblastoma occurring after the treatment for ALL had unique clinical, radiological, histological, and molecular characteristics in our three cases.

DOI： 10.1007/s10014-018-0316-1

Scopus

PubMed

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

The 2016 World Health Organization classification of tumors of the central nervous system was recently revised. Mutations in the isocitrate dehydrogenase 1 (IDH1) and IDH2 genes and chromosome 1p/19q codeletion are especially important for both the integrated diagnosis and the determination of surgical strategy. To establish a method for intraoperative molecular diagnosis, a simple, rapid method was developed for the measurement of 2-hydroxyglutarate (2-HG), a specific oncometabolite formed in the presence of IDH gene mutation, using liquid chromatography/electrospray ionization tandem mass spectrometry (LC/ESI-MS/MS). This method requires only 10 min to measure the level of 2-HG from tissue preparation to completion of examination. Using this method, the level of 2-HG was analyzed in 105 patients with diffuse infiltrating glioma, and showed that IDH mutated glioma had significantly higher level of 2-HG compared to IDH wild-type glioma. Receiver operating characteristic curve analysis showed the area under the curve, sensitivity, and specificity were 0.9815, 97.5, and 100%, respectively. In contrast, tumor grade and presence of chromosome 1p/19q codeletion in the IDH mutated glioma could not be predicted from the level of 2-HG. Measurement of 2-HG level using LC/ESI-MS/MS can provide rapid and accurate information of mutation status in the IDH gene.

DOI： 10.1007/s10014-018-0317-0

PubMed

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

Expression of CD44 in glioma cells was previously correlated with tumor grade and is considered a stem cell marker. CD44 stabilizes the cystine-glutamate transporter (xCT) and inhibits apoptosis in cancer stem cells (CSCs). Recently it was found that Sulfasalazine (SSZ), an anti-inflammatory drug, acts as an inhibitor of xCT and therefore has potential as a targeted therapy for CSCs. In this study, we tested an efficacy of SSZ against glioma stem cell model developed in rats. As poor penetration of blood-brain barrier resulted in insufficient efficacy of systemic SSZ treatment, SSZ was delivered locally with convection-enhanced delivery (CED). In vitro, expression of CD44 in glioma cells and efficacy of SSZ against glioma cells and glioma stem cells were confirmed. SSZ demonstrated anti-proliferative activity in a dose dependent manner against these cells. This activity was partially reversible with the addition of antioxidant, N-acetyl-L-cysteine, to the medium. In vivo, CED successfully delivered SSZ into the rat brain parenchyma. When delivered at 5 mM concentration, which was the highest possible concentration when SSZ was dissolved in water, CED of SSZ resulted in almost no tissue damage. Against highly malignant bRiTs-G3 brain tumor xenografted rat model; the glioma stem cell model, CED of SSZ at 5 mM concentration induced apoptosis and prolonged survival. Consequently, CED of SSZ induced glioma stem cell death without evidence of tissue damage to normal brain parenchyma. This strategy may be a promising targeted treatment against glioma stem cells.

DOI： 10.1007/s11060-017-2621-7

PubMed

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

Objective: To clarify the clinical, neuropathologic, and virologic characteristics of progressive multifocal leukoencephalopathy (PML) and its immune reconstitution inflammatory syndrome (IRIS) in a patient with fingolimod-treated MS. Methods: A case study. Results: A 34-year-old patient with MS using fingolimod for 4 years had a gradual progression of right hemiparesis and aphasia with a new subcortical white matter lesion in the precentral gyrus by initial MRI. Blood tests were normal, except for lymphopenia (160 cells/μL). One month after the cessation of fingolimod, brain MRI depicted a diffusely exacerbated hyperintensity on fluid-attenuated inversion recovery and diffusion-weighed imaging in the white matter with punctate gadolinium enhancement, suggesting PML-IRIS. A very low level of JC virus (JCV)-DNA (15 copies/mL) was detected in the CSF as judged by quantitative PCR. Brain tissues were biopsied from the left frontal lesion, which showed some small demyelinated foci with predominant loss of myelin-associated glycoprotein with infiltrations of lymphocytes and macrophages, but clear viral inclusion was not observed with hematoxylin-eosin staining. JCV-DNA was uniquely detectable in an active inflammatory demyelinating lesion by in situ hybridization, possibly suggesting an early phase of PML. DNA extracted from the brain sample was positive for JCV-DNA (151 copies/cell). It took 3 months to normalize the blood lymphocyte count. The patient was treated with 1 g of IV methylprednisolone for 3 days and a weekly oral dose (375 mg) of mefloquine, and her symptoms gradually improved. Conclusion: Low CSF JCV-DNA and unfound viral inclusions initially made her diagnosis difficult. The clinical course of fingolimod-associated PML may be associated with mild immune reconstitution.

DOI： 10.1212/NXI.0000000000000415

PubMed

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

Pilocytic astrocytomas and low-grade gliomas are more common compared with glioblastomas in patients with neurofibromatosis 1 (NF1). A recent genome-wide analysis has shown frequent NF1 gene alterations in the mesenchymal subtype of a glioblastoma; however, little is known about clinicopathological features of glioblastomas in NF1 patients (NF1 glioblastomas). We analyzed four NF1 glioblastomas. Radiographical and intraoperative findings showed well-circumscribed tumors from surrounding brain. Pathological analysis presented a paucity of processes with an eosinophilic cytoplasm, bizarre nuclei, xanthomatous-like appearance, multinucleated giant cells, and histiocytoid appearance. During the follow-up period, one patient died at 49 months and others remained alive for 60, 87, and 106 months; thus, patients with NF1 glioblastoma presented a relatively favorable survival. None of the NF1 glioblastomas harbored isocitrate dehydrogenase 1 (IDH1) gene mutation, v-RAF murine sarcoma viral oncogene homolog B1 (BRAF) gene mutation, and telomerase reverse transcriptase (TERT) gene promoter mutation. We identified that NF1 glioblastoma is a unique subset of glioblastoma.

DOI： 10.1007/s10014-017-0302-z

PubMed

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

Carmustine wafers (CW) were approved in Japan for newly diagnosed and recurrent malignant gliomas during 2013. The ventricle is often opened during surgery to achieve maximum resection. While not generally recommended in such situations, CW might be safely achieved by occluding an opened ventricle using gelform or collagen sheets. However, whether CW implantation actually confers a survival benefit for patients who undergo surgery with an open ventricle to treat glioblastoma remains unclear. Clinical, imaging, and survival data were collected in this multicenter retrospective study of 122 consecutive patients with newly diagnosed glioblastoma to determine adverse events and efficacy. Overall, 54 adverse events of all grades developed in 35 (28.6%) patients, with the most common being new seizures (16%). Adverse events did not significantly differ between patients with opened and closed ventricles during surgery. The 10- and 21.7-month, median, progression-free (PFS) and overall survival (OS), respectively did not significantly differ according to resection rates. However, median PFS and OS were significantly longer among patients with closed, than open ventricles (12.8 vs. 7.4 months; p = 0.0039 and 26.9 vs. 18.6 months; p = 0.011, respectively). Implanting CW into the resection cavity during concomitant radiochemotherapy with temozolomide seems to yield better survival rates without increased adverse events. Occlusion of the ventricular opening during surgery might be safe for CW implantation, but less so for treating patients with newly diagnosed glioblastoma.

DOI： 10.1007/s11060-017-2488-7

Web of Science

PubMed

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

The indications of surgery for thalamic high-grade gliomas are not well established. The present study investigated the outcome of 21 patients treated by surgery and reports the high incidence of distant recurrences including disseminations after successful removal. Twenty-one patients with thalamic high-grade gliomas not invading the pyramidal tract or midbrain underwent cytoreductive surgery at our institute from June 1997 to August 2015. Surgery was performed with the aid of a neuronavigation system, electrophysiological monitoring, and fluorescence navigation. Tumor histology included 12 cases of the World Health Organization grade III and nine cases of grade IV. Gross total resection was achieved in six cases, subtotal in 13, and partial in two. Motor weakness accompanied by sensory disturbance deteriorated immediately after surgery in 13 patients. However, five patients were determined to show deterioration at 2 months after surgery. Postoperative radiation and chemotherapy were given to every patient, and median progression-free survival of patients with grade III and IV tumors was 12.1 and 7.0 months, respectively. Median overall survival of patients with grade III and IV tumors was 25.6 and 12.6 months, respectively. High incidence of distant recurrences was found, with distant lesions at recurrence in 13 of 19 patients with recurrence, suggesting the life-restricting factor in these patients. Thalamic high-grade glioma without invasion into the pyramidal tract and brainstem can be considered as a candidate for surgical resection. Distant lesion limits the survival of patients after successful resection.

DOI： 10.1007/s10143-016-0804-x

Web of Science

PubMed

Language：English   Publishing type：Research paper (scientific journal)   Publisher：SPRINGER WIEN  

Convection-enhanced delivery (CED) is a technique allowing local infusion of therapeutic agents into the central nervous system, circumventing the blood-brain or spinal cord barrier.
To evaluate the utility of nimustine hydrochloride (ACNU) CED in controlling tumor progression in an experimental spinal cord glioma model.
Toxicity studies were performed in 42 rats following the administration of 4 mu l of ACNU CED into the mid-thoracic spinal cord at concentrations ranging from 0.1 to 10 mg/ml. Behavioral analyses and histological evaluations were performed to assess ACNU toxicity in the spinal cord. A survival study was performed in 32 rats following the implantation of 9 L cells into the T8 spinal cord. Seven days after the implantation, rats were assigned to four groups: ACNU CED (0.25 mg/ml; n = 8); ACNU intravenous (i.v.) (0.4 mg; n = 8); saline CED (n = 8); saline i.v. (n = 8). Hind limb movements were evaluated daily in all rats for 21 days. Tumor sizes were measured histologically.
The maximum tolerated ACNU concentration was 0.25 mg/ml. Preservation of hind limb motor function and tumor growth suppression was observed in the ACNU CED (0.25 mg/ml) and ACNU i.v. groups. Antitumor effects were more prominent in the ACNU CED group especially in behavioral analyses (P &lt; 0.05; log-rank test).
ACNU CED had efficacy in controlling tumor growth and preserving neurological function in an experimental spinal cord tumor model. ACNU CED can be a viable treatment option for spinal cord high-grade glioma.

DOI： 10.1007/s00701-017-3123-2

Web of Science

PubMed

Language：English   Publishing type：Research paper (scientific journal)   Publisher：ELSEVIER SCIENCE INC  

OBJECTIVE: We previously disclosed that damage to the subependymal arteries (SEAs) caused by coagulation of the choroid plexus at the atrium can result in infarction within the lateral posterior choroidal artery territory, followed by hemiparesis. The present study describes the intra-operative anatomical findings of the SEAs and choroid plexus at the atrium, which were verified only by a few cadaveric studies.
METHODS: Locations of the SEA and descending motor pathway were determined with the neuronavigation system and subcortical electrical stimulation in 8 cases of periatrial brain tumor. Indocyanine green videoangiography was performed to verify the blood flowin the choroid plexus and SEAs.
RESULTS: Intraoperative visualization of the SEAs was performed successfully in all patients. The neuronavigation system and subcortical electrical stimulation mapping demonstrated that these SEAs penetrated into the descending motor pathway. Indocyanine green depicted the blood flow of the SEAs entering the wall of the lateral ventricle and adjacent brain parenchyma. The blood flow directions between the SEAs and choroid plexus were not uniform, because the SEAs were filled ahead of the choroid plexus in 3 cases, whereas the choroid plexus was filled first in the other 2 cases.
CONCLUSIONS: Manipulations to the inner side of the choroid plexus at the transition from the atrium to the body of lateral ventricle can damage the SEAs. Not only coagulation of the SEAs themselves, but also coagulation of choroid plexus itself may reduce the blood flow in the SEAs, resulting in ischemic complications at descending motor pathway.

DOI： 10.1016/j.wneu.2017.02.022

Web of Science

PubMed

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

Language：English   Publishing type：Research paper (scientific journal)   Publisher：JAPAN NEUROSURGICAL SOC  

Convection-enhanced delivery (CED) circumvents the blood-brain barrier by delivering agents directly into the tumor and surrounding parenchyma. CED can achieve large volumes of distribution by continuous positive-pressure infusion. Although promising as an effective drug delivery method in concept, the administration of therapeutic agents via CED is not without challenges. Limitations of distribution remain a problem in large brains, such as those of humans. Accurate and consistent delivery of an agent is another challenge associated with CED. Similar to the difficulties caused by immunosuppressive environments associated with gliomas, there are several mechanisms that make effective local drug distribution difficult in malignant gliomas. In this review, methods for local drug application targeting gliomas are discussed with special emphasis on CED. Although early clinical trials have failed to demonstrate the efficacy of CED against gliomas, CED potentially can be a platform for translating the molecular understanding of glioblastomas achieved in the laboratory into effective clinical treatments. Several clinical studies using CED of chemotherapeutic agents are ongoing. Successful delivery of effective agents should prove the efficacy of CED in the near future.

DOI： 10.2176/nmc.ra.2016-0071

Web of Science

PubMed

Language：English   Publishing type：Research paper (scientific journal)   Publisher：JAPAN NEUROSURGICAL SOC  

Immature teratoma (IMT) is normally treated by resection and adjuvant therapy. The present unusual case of recurrent germinoma occurred 21 years after total resection of pineal IMT. A 3-year-old boy presented with headache, disturbance of consciousness, and Parinaud's syndrome. Magnetic resonance (MR) imaging revealed a pineal mass lesion, and total resection of the tumor was achieved. The histological diagnosis was mature teratoma. He did not receive further treatment, and did well without recurrence for 20 years. However, he suffered headache 21 years after resection, and MR imaging revealed a homogeneously enhanced pineal mass with low minimum apparent diffusion coefficient value and proton MR spectroscopy showed a huge lipid peak. The levels of tumor markers were not elevated. Cerebrospinal fluid (CSF) cytology found atypical cells with large nuclei and irregularly shaped nucleoli. To elucidate the relationship between the primary and recurrent tumors, we reviewed the histological specimens and CSF cytology at the initial treatment and found a subset of incompletely differentiated components resembling fetal tissues in the histological specimen and atypical large cells in the CSF. Based on these radiological and histological findings, we presume that the recurrent disease was disseminated germinoma after the resection of disseminated IMT. He received chemotherapy and craniospinal radiation therapy, and the enhanced lesion and atypical cells in the CSF disappeared. This case demonstrates that disseminated IMT can be controlled for the long term without adjuvant therapy, but may recur as germinoma. Tumor dormancy may account for this unusual course.

DOI： 10.2176/nmc.cr.2016-0241

Web of Science

PubMed

Language：English   Publisher：Japan Neurosurgical Society  

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

DOI： 10.2176/jns-nmc.2022-0056

PubMed

CiNii Research

Language：English   Publisher：The Japan Neurosurgical Society  

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

DOI： 10.2176/jns-nmc.2022-0036

PubMed

CiNii Research

Language：English   Publisher：The Japan Neurosurgical Society  

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

DOI： 10.2176/jns-nmc.2022-0055

PubMed

CiNii Research

Language：English   Publisher：Surgical Neurology International  

Background: Schwannomatosis (SWN) is genetically similar to neurofibromatosis type 2 (NF2) and represents a NF2 gene mutation. Previous studies have shown that these mutations in both neurons and Schwann cells can lead to the development of schwannomas after nerve crush injuries. Here, we reviewed the potential pathoanatomical mechanisms for the development of a trauma-induced spinal schwannomas in a 55-year-old male with SWN. Case Description: A 49-year-old male had originally undergone a L3-L5 lumbar laminectomy for stenosis; the schwannomas seen on the preoperative magnetic resonance imaging (MRI) were not resected. Now at age 55, he newly presented with low back pain and numbness in the left L5 dermatome, and he was diagnosed with an L4 vertebral level cauda equina tumor on MRI. Following gross-total resection, the histopathological assessment revealed a Ki-67 labeling index 5-10% in hotspots (i.e., slightly higher than the normal range of schwannomas) and a 20% mosaic loss of SMARCB1. Based on these criteria, he was diagnosed as having SWN. Conclusion: In this patient with SWN, compression/physical trauma to nerves of the cauda equina during the L3-L5 laminectomy 6 years ago likely caused the progression of schwannoma.

DOI： 10.25259/SNI_453_2022

Scopus

PubMed

Language：English   Publisher：The Japan Neurosurgical Society  

<p>Spinal cord parenchymal lesions may induce intractable neuropathic pain. However, the efficacy of conventional spinal cord stimulation for the neuropathic pain following spinal cord lesions remains to be controversial. In this study, we present three cases of spinal cord stimulation using a paddle lead at the rostral side of the spinal lesion causing pain symptoms. Good pain reductions were achieved using conventional stimulation in one case and using differential target multiplexed stimulation in two cases. Case 1: A 55-year-old man presented with neuropathic pain affecting his bilateral upper extremities due to a traumatic cervical spinal cord injury. Conventional stimulation via a paddle-type electrode was able to reduce the pain from 8 to 4 via a visual analog scale. Case 2: A 67-year-old man had undergone three spinal surgeries. He presented with pain and numbness of bilateral lower extremities due to a spinal cord lesion by thoracic disc herniation. Differential target multiplexed stimulation via a paddle-type electrode achieved excellent pain reduction, that is, from 9 to 2 on the visual analog scale. Case 3: An 80-year-old man presented with pain in his bilateral upper extremities due to a cervical spinal cord lesion caused by compression and spinal canal stenosis. Posterior cervical decompression and paddle-type electrode placement were performed simultaneously. Differential target multiplexed stimulation was able to achieve excellent pain reduction, from 7 to 2 on the visual analog scale. Spinal cord stimulation using a paddle lead at the rostral side of the spinal lesion and differential target multiplexed stimulation may provide significant opportunities for patients with intractable neuropathic pain following spinal cord lesions.</p>

DOI： 10.2176/jns-nmc.2022-0218

PubMed

CiNii Research

Language：English   Publisher：Journal of Spine Surgery  

Solitary fibrous tumors (SFTs) and hemangiopericytomas (HPCs) have been classified as one entity by the World Health Organization in 2016 due to gene fusion between NAB2 and STAT6. In the Central Nervous System (CNS), a hypocellular, collagenized tumor with a classic SFT phenotype is considered grade I, whereas more densely cellular tumors mostly corresponding to the HPC phenotype are classified as grade II or III (anaplastic) depending in mitotic count (<5 vs. >5 mitoses per 10 high-power fields). Herein, we report two cases of targeted SFT/HPC in which pathological differences and WHO grading affect clinical features. A 75-year-old woman presented with headache and had an intradural extramedullary tumor at the C1 to C2 level. The tumor was well-circumscribed and attached only to the dura mater. It was totally removed and diagnosed SFT/HPC grade I. In contrast, a 68-year-old woman presented with numbness in the right upper limb and had an intradural extramedullary tumor at the medulla to C3 levels The tumor was irregularly marginated and strongly adherent to the spinal cord and involved the vertebral artery. It was sub totally removed and diagnosed SFT/HPC grade II. To the best of our knowledge, there are only 12 cases of SFT/HPC at the craniocervical junction, including the present two cases, of which four that were adherent to the spinal cord or involved the vertebral artery were grade II or III. Although the location of the tumor was almost the same, there were significant differences in the intraoperative findings according to the WHO grading.

DOI： 10.21037/jss-21-83

Scopus

PubMed

Language：English   Publisher：The Japan Neurosurgical Society  

<p>Iatrogenic vertebral artery (VA) injury in cervical fusion is an extremely rare complication but can lead to serious sequelae. We present two successful cases of internal trapping for preventing delayed-onset ischemic stroke after iatrogenic VA stenosis caused by a cervical pedicle screw. A 34-year-old female underwent posterior cervical fusion for C4/C5 dislocation fracture. No neurological deficits were observed after the operation. However, the postoperative images revealed that the left C5 pedicle screw perforated the transverse foramen, and the left VA was suspected to be occluded at the screw insertion site. Before revision surgery, we tried to embolize the injured VA with coils. A microcatheter could be navigated from the ipsilateral VA to the distal of the screw, and internal trapping was performed with coils. Another case is that of a 50-year-old male with cervical spondylosis, who underwent posterior decompression and cervical fusion. The neurological symptoms did not deteriorate after the operation. However, the postoperative computed tomography images revealed the perforation of the right C3 transverse foramen by the pedicle screw. In right vertebral angiography, about 70% stenosis was observed at the screw insertion site. Although revision surgery was not planned due to good stability, we embolized the right VA after balloon occlusion test, to prevent the delayed-onset thromboembolic complications. Both the patients recovered without any neurological deficits. Iatrogenic VA injuries, even if asymptomatic immediately after surgery, can lead to serious sequelae in case of delayed-onset ischemic stroke. Therefore, careful attention should be paid when the screw perforates the transverse foramen.</p>

DOI： 10.2176/nmccrj.cr.2021-0062

PubMed

CiNii Research

J-GLOBAL

J-GLOBAL

Language：English   Publisher：The Japan Neurosurgical Society  

<p>Glioblastoma multiforme (GBM) is an aggressive cancer type, with fewer than 3–5% of patients surviving for more than 3 years. We describe a 48-year-old right-handed man who presented with generalized seizure attacks. Magnetic resonance imaging (MRI) revealed a heterogeneous gadolinium-enhancing lesion in the left inferior parietal lobule. The patient underwent awake surgery, and tumor resection included abnormalities on T2-weighted MRI, with subcortical mapping used to identify the deep functional boundaries. After supratotal resection, the tumor was diagnosed as GBM without isocitrate dehydrogenase (<i>IDH</i>) 1 and 2 mutations. At a follow-up evaluation, 9 years and 2 months after the surgery, the patient appeared healthy, and no relapse or recurrence was observed. We present the case of a long-term survivor of <i>IDH</i>-wildtype GBM. This case suggests that supratotal resection with intraoperative awake brain mapping can improve survival without impairing the patient’s neurological functions.</p>

DOI： 10.2176/nmccrj.cr.2021-0120

PubMed

CiNii Research

Language：English   Publisher：The Japan Neurosurgical Society  

<p>Intramedullary spinal cord tumors are rare in children. Regardless of the type of tumor, surgical removal is thought to improve progression-free survival. However, postoperative kyphosis is a serious problem in children, who can expect long-term survival. We present a pediatric case of neurofibromatosis type 2-related spinal ependymoma at the cervicothoracic regions where acute neurological deterioration was developed due to a combination of tumor recurrence and postoperative kyphotic deformity. In the first surgery, subtotal tumor resection was performed via osteoplastic laminotomy. Postoperative radiological evaluation at several months showed cervicothoracic junctional kyphosis, which subsequently made a significant improvement by lifestyle instructions. However, 22 months after the surgery, he exhibited rapid neurological deterioration caused by the regrowth of the recurrent tumor and re-emergence of kyphotic deformity, which led to the fixed laminar flap sank into the spinal canal. Therefore, a second surgery was performed 23 months after the first surgery, and gross total removal was achieved. Osteoplastic laminotomy is presumed to reduce the occurrence of postoperative kyphosis compared with laminectomy, but there have been no reports on the spinal cord compression by plunging of the re-fixed laminar flap into the spinal canal. The kyphosis deformity increases the chance of re-fixed laminar flap coming off, thereby accelerating neurological injury on top of the neural damage by tumor recurrence itself. Therefore, pediatric patients with spinal cord tumors should be carefully managed in terms of recurrent tumors and postoperative kyphosis, and timely surgical intervention is necessary before kyphotic deformity becomes evident.</p>

DOI： 10.2176/nmccrj.cr.2021-0086

PubMed

CiNii Research

J-GLOBAL

Language：English   Publishing type：Research paper, summary (international conference)   Publisher：OXFORD UNIV PRESS INC  

Web of Science

Language：Japanese   Publisher：(NPO)日本脳神経血管内治療学会  

Language：English   Publishing type：Research paper, summary (international conference)   Publisher：AMER SOC CLINICAL ONCOLOGY  

DOI： 10.1200/JCO.2019.37.15_suppl.2004

Web of Science

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

Language：English   Publishing type：Research paper, summary (international conference)   Publisher：SAGE PUBLICATIONS LTD  

Web of Science

Language：Japanese   Publisher：(公社)日本小児科学会  

Language：English   Publishing type：Research paper, summary (international conference)   Publisher：OXFORD UNIV PRESS INC  

Web of Science

Language：English   Publishing type：Research paper, summary (international conference)   Publisher：OXFORD UNIV PRESS INC  

Web of Science

Language：English   Publishing type：Research paper, summary (international conference)   Publisher：OXFORD UNIV PRESS INC  

Web of Science

Language：Japanese   Publisher：(公社)日本小児科学会  

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

Language：English   Publishing type：Research paper, summary (international conference)   Publisher：SAGE PUBLICATIONS LTD  

Web of Science

Event date： 2021.10

Language：Japanese   Presentation type：Oral presentation (invited, special)  

Venue：横浜  

Event date： 2021.10

Language：Japanese   Presentation type：Symposium, workshop panel (nominated)  

Venue：横浜  

Event date： 2021.9

Language：Japanese   Presentation type：Oral presentation (invited, special)  

Venue：名古屋  

Event date： 2021.5

Language：Japanese   Presentation type：Symposium, workshop panel (nominated)  

Venue：大阪  

Event date： 2021.4

Language：Japanese   Presentation type：Symposium, workshop panel (nominated)  

Venue：札幌  

Event date： 2021.4

Language：Japanese   Presentation type：Oral presentation (invited, special)  

Venue：金沢  

Event date： 2021.3

Language：Japanese   Presentation type：Oral presentation (invited, special)  

Venue：名古屋