A study of prognostic factors in 45 cases of atypical meningioma.

BACKGROUND: Atypical meningioma differs from Grade I meningioma in terms of high recurrence rate and short life expectancy. We evaluated the clinical course of atypical meningioma and investigated prognostic factors affecting its outcomes. METHOD: We reviewed 45 patients with atypical meningioma who underwent surgical intervention between January 2000 and December 2013. The mean age of the patients and mean follow-up period was 58.7 years and 81.0 months, respectively. Analyses included factors such as patient age, gender, location and size of tumor, extent of surgical resection (Simpson Grading System), and MIB-1 labeling index (LI). Univariate analysis was used to detect prognostic factors associated with recurrence and survival. RESULTS: The 5-year recurrence-free rate for all 45 patients was 58.4 %; 5- and 10-year survival rates were 83.2 % and 79.9 %, respectively. In univariate analyses, age >60 years, and MIB-1 LI correlated with disease recurrence, whereas age >60 years, subtotal surgical resection, MIB-1 LI, and indication for radiotherapy correlated with death. MIB-1 LI levels higher than 12.8 % and 19.7 % predicted recurrence and death, respectively. In our cohort, 26 patients received postoperative radiotherapy including conventional radiation (n = 21) or gamma knife radiosurgery (n = 5). Postoperative radiotherapy did not decrease recurrence rates in our cohort (p = 0.63). Six and two patients who died during the study period underwent conventional radiation and radiosurgery, respectively. CONCLUSIONS: Age, male gender, extent of surgical resection, and higher MIB-1 LI influenced the outcome of atypical meningioma. In our cohort, postoperative radiotherapy failed to provide long-term tumor control. Following incomplete surgical resection of atypical meningioma in elderly patients, adjuvant postoperative radiotherapy may not be an ideal treatment option, particularly when MIB-1 LI is higher than 19.7 %.

Mutations in genes encoding the glycine cleavage system predispose to neural tube defects in mice and humans.

Neural tube defects (NTDs), including spina bifida and anencephaly, are common birth defects of the central nervous system. The complex multigenic causation of human NTDs, together with the large number of possible candidate genes, has hampered efforts to delineate their molecular basis. Function of folate one-carbon metabolism (FOCM) has been implicated as a key determinant of susceptibility to NTDs. The glycine cleavage system (GCS) is a multi-enzyme component of mitochondrial folate metabolism, and GCS-encoding genes therefore represent candidates for involvement in NTDs. To investigate this possibility, we sequenced the coding regions of the GCS genes: AMT, GCSH and GLDC in NTD patients and controls. Two unique non-synonymous changes were identified in the AMT gene that were absent from controls. We also identified a splice acceptor site mutation and five different non-synonymous variants in GLDC, which were found to significantly impair enzymatic activity and represent putative causative mutations. In order to functionally test the requirement for GCS activity in neural tube closure, we generated mice that lack GCS activity, through mutation of AMT. Homozygous Amt(-/-) mice developed NTDs at high frequency. Although these NTDs were not preventable by supplemental folic acid, there was a partial rescue by methionine. Overall, our findings suggest that loss-of-function mutations in GCS genes predispose to NTDs in mice and humans. These data highlight the importance of adequate function of mitochondrial folate metabolism in neural tube closure.

Symptomatic vasospasm in elderly patients with aneurysmal subarachnoid hemorrhage: comparison with nonelderly patients.

OBJECTIVE: To clarify the influence of age on the occurrence of symptomatic vasospasm (SVS), we retrospectively compared 34 elderly (over 70 years) and 71 nonelderly patients with aneurysmal subarachnoid hemorrhage (SAH). METHODS: Between 2008 and 2010, at our hospital 105 patients (Hunt and Kosnik grades I-IV) underwent aneurysm surgery within 72 h of the insult. They were divided into four groups based on their age (younger/older than 70 years) and treatment (aneurysmal clipping or coiling). In all patients, we used the same protocol, which included the delivery of intrathecal urokinase and intravenous fasudil chloride; in patients with angiographic evidence of vasospasm, we also injected fasudil chloride intra-arterially. RESULTS: Among the elderly patients, 4.3% of those treated by clipping and 9.1% of those treated by coiling experienced SVS; the comparative incidence in younger patients was 6.5% and 4.0%, respectively. The differences were not statistically significant (p = 0.40). The ratio of ventriculo peritoneal (VP) shunts was higher in the elderly patients (p = 0.00007). The incidence of favorable treatment outcomes was significantly lower in elderly patients (p = 0.00004). CONCLUSION: Under our treatment protocol, patient age did not affect the incidence of SVS. Our protocol may be effective for the prevention of SVS after aneurysmal SAH regardless of patient age.

Cerebral infarction following administration of andexanet alfa for anticoagulant reversal in a patient with traumatic acute subdural hematoma.

Background: Anticoagulants prevent thrombosis in patients with atrial fibrillation (AF) and venous thromboembolism but increase the risk of hemorrhagic complications. If severe bleeding occurs with anticoagulant use, discontinuation and rapid reversal are essential. However, the optimal timing for resuming anticoagulants after using reversal agents remains unclear. Here, we report early cerebral infarction following the use of andexanet alfa (AA), a specific reversal agent for factor Xa inhibitors, in a patient with traumatic acute subdural hematoma (ASDH). The possible causes of thromboembolic complication and the optimal timing for anticoagulant resumption are discussed. Case Description: An 84-year-old woman receiving rivaroxaban for AF presented with impaired consciousness after a head injury. Computed tomography (CT) revealed right ASDH. The patient was administered AA and underwent craniotomy. Although the hematoma was entirely removed, she developed multiple cerebral infarctions 10 h after the surgery. These infarctions were considered cardiogenic cerebral embolisms and rivaroxaban was therefore resumed on the same day. This case indicates the possibility of early cerebral infarction after using a specific reversal agent for factor Xa inhibitors. Conclusion: Most studies suggest that the safest time for resuming anticoagulants after using reversal agents is between 7 and 12 days. The present case showed that embolic complications may develop much earlier than expected. Early readministration of anticoagulant may allow for adequate prevention of the acute thrombotic syndromes.

Incidence of Chronic Subdural Hematoma after Mild Head Trauma in Elderly Patients with or without Pre-traumatic Conditioning of Anti-thrombotic Drugs.

Anti-thrombotic drugs may increase the risk for chronic subdural hematoma (CSDH). However, whether to continue or discontinue/counteract these drugs has not been investigated in patients with mild head trauma. CSDH incidence after mild head trauma, as well as the risk for CSDH in patients with anti-thrombotic drugs, were investigated in this study. The study included 765 consecutive elderly (>65 y.o.) patients with mild head trauma and an initial Glasgow Coma Scale (GCS) score of 14 or 15. All patients received initial CT within 24 hours after trauma and were re-examined 30 days after trauma to detect CSDH formation, repeating for every 30 days to examine symptomatic CSDH progression. Patients were divided into two groups, with anti-thrombotic drugs (n = 195) or without them (n = 263), to investigate the influence of pre-traumatic conditioning with anti-thrombotic drugs on CSDH. The whole sample was 458 out of 765 cases. The incidence of CSDH formation was 91 out of 458 cases (19.9%) after mild head trauma, with no significant difference between with and without anti-thrombotic drugs. CSDH progressed as symptomatic in 21 out of 458 cases (4.6%), with no significant difference between with and without anti-thrombotic drugs. Pre-traumatic conditioning with anti-thrombotic drugs and its continuation after trauma did not affect the incidence of formation or symptomatic progression of CSDH. This finding suggests that discontinuing and/or counteracting anti-thrombotic drugs may be unnecessary in patients with mild head trauma.

Correlations of intracranial pathology and cause of head injury with retinal hemorrhage in infants and toddlers: A multicenter, retrospective study by the J-HITs (Japanese Head injury of Infants and Toddlers study) group.

INTRODUCTION: In infants who have suffered head trauma there are two possible explanations for retinal hemorrhage (RH): direct vitreous shaking and occurrence in association with intracranial lesions. Which possibility is more plausible was examined. MATERIAL AND METHODS: This multicenter, retrospective study reviewed the clinical records of children younger than four years with head trauma who had been diagnosed with any findings on head computed tomography (CT) and/or magnetic resonance imaging (MRI). Of 452 cases, 239 underwent an ophthalmological examination and were included in this study. The relationships of RH with intracranial findings and the cause of injury were examined. RESULT: Odds ratios for RH were significant for subdural hematoma (OR 23.41, p = 0.0004), brain edema (OR 5.46, p = 0.0095), nonaccidental (OR 11.26, p<0.0001), and self-inflicted falls (OR 6.22, p = 0.0041). CONCLUSION: Although nonaccidental, brain edema and self-inflicted falls were associated with RH, subdural hematoma was most strongly associated with RH.

[Operated case of a ruptured aneurysm arising from fenestration of the vertebral artery].

Aneurysm arising from duplication of the vertebral artery (VA) is rare. We report a surgical case of an aneurysm of VA fenestration and provide a literature review. A 42-year-old man suffered from sudden onset of severe headache. CT image revealed subarachnoid hemorrhage predominantly in the posterior fossa. Digital subtraction angiography showed a fenestration of the right VA and the aneurysm arising from the proximal limb of the fenestration. Neck clipping of the aneurysm was performed because embolization was difficult due to the wide neck. The patient was discharged without deficits. Seven cases of an aneurysm of the VA fenestration have been reported. Five cases underwent coil embolization, and 2 underwent craniotomy, trapping, and resection. This is the first case of neck clipping of an aneurysm of the VA fenestration.

Unexplained mechanism of subdural hematoma with convulsion suggests nonaccidental head trauma: A multicenter, retrospective study by the Japanese Head injury of Infants and Toddlers study (J-HITs) group.

OBJECTIVE: The medical history of injury given by parents of infants and toddlers with head trauma may not be accurate or completely true. The purpose of this study was to examine the relationship between subdural hematoma (SDH) due to nonaccidental injury and mechanisms of injury provided by caregivers. METHODS: Our multicenter study group retrospectively reviewed the clinical records of children younger than 4 years with head trauma who have been diagnosed with any finding on head computed tomography (CT) and/or magnetic resonance imaging (MRI). A total of 84 cases of subdural hematomas with retinal findings, including cases reported to the child guidance center and traffic and birth injuries, were included in the study. They were classified by the mechanism of injury provided by the caregivers. Clinical findings were reviewed and classified into nonaccidental and accidental groups. The mechanisms of the injuries were examined by multivariable analysis to identify which ones were statistically associated with nonaccidental injuries. RESULTS: Of the 84 patients with SDHs, 51 were classified into the nonaccidental group, and 33 children were classified into the accidental group. In 19 patients with a chief complaint of convulsion who had SDH but no episode of trauma, 18 were classified into the nonaccidental group. On multivariable analysis, unexplained convulsions (odds ratio: 12.04, 95% confidence interval: 1.44-100.49) were significantly associated with increased odds of nonaccidental injury. CONCLUSIONS: In the present study, there was a relationship between nonaccidental injury and unexplained SDH with a chief complaint of convulsion.

Infantile subdural hematoma in Japan: A multicenter, retrospective study by the J-HITs (Japanese head injury of infants and toddlers study) group.

OBJECTIVE: Subdural hematoma in infants or toddlers has often been linked to abuse, but it is not clear how many cases actually occur and how many are suspected of abuse. The purpose of this study was to investigate subdural hematoma in infants and toddlers in Japan. METHODS: This multicenter, retrospective study reviewed the clinical records of children younger than 4 years with head trauma who were diagnosed with any finding on head computed tomography (CT) and/or magnetic resonance imaging (MRI), such as skull fracture and/or intracranial injury. A total of 452 children were included. The group suspected to have been abused was classified as nonaccidental, and the group considered to have been caused by an accident was classified as accidental. Subdural hematoma and other factors were examined on multivariate analysis to identify which factors increase the risk of nonaccidental injuries. RESULTS: Of the 452 patients, 158 were diagnosed with subdural hematoma. Subdural hematoma was the most common finding intracranial finding in head trauma in infants and toddlers. A total of 51 patients were classified into the nonaccidental group, and 107 patients were classified into the accidental group. The age of patients with subdural hematoma showed a bimodal pattern. The mean age of the accidental group with subdural hematoma was significantly older than that in the nonaccidental group (10.2 months vs 5.9 months, respectively. p < 0.001). Multivariate analysis showed that patients under 5 months old, retinal hemorrhage, and seizure were significant risk factors for nonaccidental injury (odds ratio (OR) 3.86, p = 0.0011; OR 7.63, p < 0.001; OR 2.49, p = 0.03; respectively). On the other hand, the odds ratio for subdural hematoma was 1.96, and no significant difference was observed (p = 0.34). CONCLUSIONS: At least in Japanese children, infantile subdural hematoma was frequently observed not only in nonaccidental but also in accidental injuries. In infants with head trauma, age, the presence of retinal hemorrhage, and the presence of seizures should be considered when determining whether they were abused. Subdural hematoma is also a powerful finding to detect abuse, but care should be taken because, in some ethnic groups, such as the Japanese, there are many accidental cases.

A genome-wide association study identifies RNF213 as the first Moyamoya disease gene.

Moyamoya disease (MMD) shows progressive cerebral angiopathy characterized by bilateral internal carotid artery stenosis and abnormal collateral vessels. Although ∼ 15% of MMD cases are familial, the MMD gene(s) remain unknown. A genome-wide association study of 785,720 single-nucleotide polymorphisms (SNPs) was performed, comparing 72 Japanese MMD patients with 45 Japanese controls and resulting in a strong association of chromosome 17q25-ter with MMD risk. This result was further confirmed by a locus-specific association study using 335 SNPs in the 17q25-ter region. A single haplotype consisting of seven SNPs at the RNF213 locus was tightly associated with MMD (P = 5.3 × 10(-10)). RNF213 encodes a really interesting new gene finger protein with an AAA ATPase domain and is abundantly expressed in spleen and leukocytes. An RNA in situ hybridization analysis of mouse tissues indicated that mature lymphocytes express higher levels of Rnf213 mRNA than their immature counterparts. Mutational analysis of RNF213 revealed a founder mutation, p.R4859K, in 95% of MMD families, 73% of non-familial MMD cases and 1.4% of controls; this mutation greatly increases the risk of MMD (P = 1.2 × 10(-43), odds ratio = 190.8, 95% confidence interval = 71.7-507.9). Three additional missense mutations were identified in the p.R4859K-negative patients. These results indicate that RNF213 is the first identified susceptibility gene for MMD.

[A case of gastric cancer accompanied by disseminated carcinomatosis of bone marrow with DIC, and subdural hematoma successfully treated with sequential methotrexate and 5-fluorouracil therapy].

A 44-year-old man visited a nearby hospital because of severe headache. Brain MRI revealed a subdural hematoma, and he was transferred to the Department of Neurosurgery of our hospital. Burr hole surgery was performed on the second day of hospitalization because of an enlargement of the hematoma. Laboratory data on admission showed the presence of a disseminated intravascular coagulation(DIC). Bone marrow aspiration revealed metastases of signet ring cell carcinoma, and abdominalCT showed gastric cancer. He was diagnosed as having DIC with bone marrow metastases of advanced gastric cancer. Despite anti-DIC therapy and blood transfusion, his systemic bleeding tendency was not improved. The neurosurgeon therefore consulted with a palliative care team. Since the patient was still young, we considered that he should be treated with anti-cancer drugs. At first, his family did not accept chemotherapy because they were pessimistic about his prognosis. However, after he regained his consciousness, we were able to perform sequential MTX and 5-FU therapy with the consent of the patient and his family. The therapy was successful, and he recovered from DIC and was discharged on the 57th hospital day.

Trans-Cell Technique through Mesh of Pipeline Embolization Device: A Case Report.

Objective: We report a case of coil embolization using trans-cell technique through mesh of a pipeline embolization device (PED). Case Presentation: A 55-year-old female developed a left cavernous carotid aneurysm (CCA) with left abducens nerve palsy. The abducens nerve palsy improved gradually after PED deployment for the aneurysm. Sixty-nine days after the procedure, the patient suddenly presented with a severe headache, left abducens nerve palsy, left eyelid edema, and left pulsatile tinnitus. Digital subtraction angiography (DSA) revealed left direct carotid cavernous fistula (dCCF) due to rupture of the aneurysm, and the patient underwent endovascular treatment. A Marathon was guided into the left internal carotid artery, and a guidewire via the Marathon passed through the mesh of the PED. Then the Marathon advanced over the guidewire into the aneurysm through the mesh of the PED, with assistance of a distal access catheter and a balloon catheter. Transarterial intra-aneurysmal coil embolization using trans-cell technique was performed, and the shunt blood flow was diminished. After subsequent transvenous embolization (TVE), the shunt blood flow disappeared, and all neurological symptoms improved. When PED is deployed linearly at a diameter 0.5 mm smaller than the nominal diameter, the average strand spacing is calculated to be approximately 0.2 mm. Since PED is a braided stent, the spacing can be large. It is theoretically reasonable for Marathon with an outer diameter of 0.59 mm to pass through the mesh of the PED. Conclusion: In some cases, trans-cell technique through mesh of PED can be performed using a small diameter microcatheter.

[A case of repeated intracerebral hemorrhage due to a ruptured aneurysm of collateral circulation from the posterior cerebral artery to the middle cerebral artery].

We present a case of ruptured distal middle cerebral artery aneurysm associated with occlusion of the anterior trunk of the ipsilateral middle cerebral artery. An 83-year-old man had right homonymous hemianopsia and right mild hemiparesis. CT scan revealed hematoma and a perifocal low density area at the left occipital subcortex without subarchnoid hemorrhage (SAH). MRI showed repeated bleeding at the same location as the CT scan. Moreover, cerebral angiograms disclosed an aneurysm on the leptomeningeal anastomosis from the left posterior cerebral artery to the middle cerebral artery. The anterior trunk of the left middle cerebral artery was occluded at the origin. The aneurysm was not situated at the bifurcation and was trapped without new neurological complications. This case suggested that hemodynamic stress due to occlusion of the main artery contributed to the development of an aneurysm at the collateral artery. Even if intraparenchymal hematoma presents without SAH, and even if it is located at a rare site for aneurysms, we should inspect the source of bleeding considering factors preventing SAH arising from aneurysms.

Mutism resulting from heterochronic bilateral cerebellar hemorrhages - A case report.

BACKGROUND: Cerebellar mutism (CM) is a neurological condition characterized by lack of speech due to cerebellar lesions. Interruption of the bilateral dentatothalamocortical (DTC) pathways at midline structure seems the principal cause of CM but not fully understood. We described a rare case of CM due to heterochronic bilateral cerebellar hemorrhages. CASE DESCRIPTION: An 87-year-old woman presented with depression of alertness after sudden vomiting. Neurologically, mild dysmetria and mutism were observed. The head computed tomography (CT) showed both a fresh right cerebellar hemorrhage and an obsolete left one. The patient was diagnosed as CM since both the thalamus and the supplementary motor area were bilaterally intact on both CT and magnetic resonance imaging. Medical treatment and rehabilitation improved her ataxia and ambulation. She became cognitively alert and could communicate by nodding, shaking her head, or facial expression. However, her mutism did not change at 4 months after the stroke. CONCLUSION: There are few reports on CM due to direct injuries to the bilateral dentate nuclei. Since our case did not show any injury other than bilateral dentate nuclei, this report can support the hypothesis that the interruptions of the bilateral DTC are the cause of CM.

Endoscopic Hematoma Evacuation for Intracerebral Hemorrhage Under Local Anesthesia: Factors That Affect the Hematoma Removal Rate.

OBJECTIVE: Recent advances in endoscopic surgery have led to more patients being able to undergo endoscopic removal of hypertensive intracerebral hemorrhage (HICH). However, because of the minimal invasiveness, endoscopic HICH removal through a narrow surgical window can result in a low removal rate. The goal of the present study was to investigate the factors that affect the removal rate of HICH evacuation. METHODS: The data from 28 patients with supratentorial HICH who had undergone endoscopic hematoma evacuation were retrospectively analyzed. The inclusion criteria were spontaneous supratentorial HICH with a hematoma volume >30 mL, admission to the hospital within 24 hours of ictus, and a Glasgow coma scale score of ≥4. RESULTS: Of the 28 patients, 9 were women and 19 were men, ranging in age from 41 to 86 years (mean, 60.7 ± 12.7). The hematoma location was the basal ganglia in 25 patients and subcortical in 3 patients. The mean preoperative hematoma volume was 62.4 ± 22.5 mL. The hematoma removal rate was <60% for 11 patients (poor evacuation group) and ≥60% for in 17 patients (good evacuation group). Comparing the 2 groups, chronic renal failure treated with hemodialysis (P = 0.0072, χ2 test), liver cirrhosis (P = 0.023, χ2 test), and surgeon experience with ≥10 cases of endoscopic HICH removal (P = 0.016, χ2 test) were significant factors related to the HICH removal rate. CONCLUSION: To achieve a good removal rate, surgeons should have experience performing the endoscopic procedure. Also, patients with end-stage chronic renal failure or liver cirrhosis should be excluded.

Model mice for mild-form glycine encephalopathy: behavioral and biochemical characterizations and efficacy of antagonists for the glycine binding site of N-methyl D-aspartate receptor.

Glycine encephalopathy (GE) is caused by an inherited deficiency of the glycine cleavage system (GCS) and characterized by accumulation of glycine in body fluids and various neurologic symptoms. Coma and convulsions develop in neonates in typical GE while psychomotor retardation and behavioral abnormalities in infancy and childhood are observed in mild GE. Recently, we have established a transgenic mouse line (low-GCS) with reduced GCS activity (29% of wild-type (WT) C57BL/6) and accumulation of glycine in the brain (Stroke, 2007; 38:2157). The purpose of the present study is to characterize behavioral features of the low-GCS mouse as a model of mild GE. Two other transgenic mouse lines were also analyzed: high-GCS mice with elevated GCS activity and low-GCS-2 mice with reduced GCS activity. As compared with controls, low-GCS mice manifested increased seizure susceptibility, aggressiveness and anxiety-like activity, which resembled abnormal behaviors reported in mild GE, whereas high-GCS mice were less sensitive to seizures, hypoactive and less anxious. Antagonists for the glycine-binding site of the N-methyl-D-aspartate receptor significantly ameliorated elevated locomotor activity and seizure susceptibility in the low-GCS mice. Our results suggest the usefulness of low-GCS mice as a mouse model for mild GE and a novel therapeutic strategy.

Genomic deletion within GLDC is a major cause of non-ketotic hyperglycinaemia.

BACKGROUND: Non-ketotic hyperglycinaemia (NKH) is an inborn error of metabolism characterised by accumulation of glycine in body fluids and various neurological symptoms. NKH is caused by deficiency of the glycine cleavage multienzyme system with three specific components encoded by GLDC, AMT and GCSH. Most patients are deficient of the enzymatic activity of glycine decarboxylase, which is encoded by GLDC. Our recent study has suggested that there are a considerable number of GLDC mutations which are not identified by the standard exon-sequencing method. METHODS: A screening system for GLDC deletions by multiplex ligation-dependent probe amplification (MLPA) has been developed. Two distinct cohorts of patients with typical NKH were screened by this METHOD: the first cohort consisted of 45 families with no identified AMT or GCSH mutations, and the second cohort was comprised of 20 patients from the UK who were not prescreened for AMT mutations. RESULTS: GLDC deletions were identified in 16 of 90 alleles (18%) in the first cohort and in 9 of 40 alleles (22.5%) in the second cohort. 14 different types of deletions of various lengths were identified, including one allele where all 25 exons were missing. Flanking sequences of interstitial deletions in five patients were determined, and Alu-mediated recombination was identified in three of five patients. CONCLUSIONS: GLDC deletions are a significant cause of NKH, and the MLPA analysis is a valuable first-line screening for NKH genetic testing.

Non-contiguous Meningeal Recurrence of Olfactory Neuroblastoma: A Case Report and Literature Review.

Olfactory neuroblastoma is an uncommon malignant tumor of neural crest origin arising from the olfactory epithelium of the superior nasal cavity. There are some reports of local recurrence or continuous extension along the olfactory epithelium to the central nervous system, but non-contiguous distant meningeal metastasis without local recurrence at the primary site is rare. We report a case of non-contiguous meningeal recurrence of olfactory neuroblastoma presenting as a giant frontal mass. A 66-year-old woman was admitted with a left nasal intranasal localized tumor without cranial extension and gross total removal was achieved. Pathological examination showed olfactory neuroblastoma and radiation therapy was added in a limited region of the removal cavity. Radiological follow-up continued for 10 years and there was no local recurrence. Sixteen years after radiation therapy, the patient found a slight frontal mass gradually growing. Magnetic resonance imaging revealed an enhanced mass lesion of 7 cm in thickness and 9 cm in diameter associated with marked thickness of the frontal bone, intradural cystic mass compressing the bilateral frontal lobe, and no local recurrence. A second operation was performed followed by radiotherapy and we diagnosed no-contiguous meningeal recurrence of metastatic olfactory neuroblastoma. Olfactory neuroblastoma is a locally aggressive tumor. Although metastasis of this tumor has been reported, non-contiguous spread to the dura is rare. Understanding the route of remote metastasis and careful evaluation after primary treatment are needed to avoid misdiagnosis and treatment delays.