Publications by authors named "Nobuya Fujita"

52 Publications

[Herpes simplex encephalitis presenting as a stroke-like episode following a migraine attack: a case report].

Rinsho Shinkeigaku 2022 Jul 24;62(7):567-570. Epub 2022 Jun 24.

Department of Neurology, Nagaoka Red Cross Hospital.

A 23-year-old woman, who had been suffering from migraine since primary school age, presented with left arm paralysis three days after one such migraine attack. On admission, brain MRI diffusion-weighted imaging (DWI) demonstrated high-signal-intensity lesions in the white matter of the right fronto-parietal lobe, and no abnormal lesions were evident in the limbic system. Although the patient had a fever of 38.7°C, the CSF cell count was not elevated. On the 4‍ day, the left arm paralysis worsened, with an increase in body temperature to 39.8°C. Brain MRI revealed that the white matter lesions had spread to the right postcentral gyrus and the bilateral insular cortex. Also, MR angiography demonstrated no spasms or dissection of the major vessels. On the 6‍ day, the CSF cell count was elevated to 54/μl and herpes simplex virus DNA was detected. Acyclovir and steroid pulse therapy ameliorated the symptoms. Cervical artery dissection and reversible cerebral vasoconstriction are well known complications of migraine attack. However, herpes simplex encephalitis should also be considered as a differential diagnosis in patients with a high fever of unknown origin.
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http://dx.doi.org/10.5692/clinicalneurol.cn-001745DOI Listing
July 2022

[A case of intravascular large B-cell lymphoma manifested as lacunar infarction].

Rinsho Shinkeigaku 2022 Jun 28;62(6):492-495. Epub 2022 May 28.

Department of Neurology, Nagaoka Red Cross Hospital.

A 79-year-old man was admitted to our hospital because of sudden onset of left ataxic hemiparesis. Brain MRI diffusion weighted images showed typical lacunar infarction on the right internal capsule. He had no risk factors of cerebrovascular disorder such as hypertension, diabetes mellitus, hyperlipidemia and arrhythmia. On admission, he had a slight fever and his laboratory data showed anemia, thrombocytopenia and elevation of CRP and LDH. Intravascular large B-cell lymphoma (IVLBCL) was suspected because the serum level of soluble IL-2 receptor was also elevated. Pathological diagnosis of IVLBCL was underwent by the skin biopsy from his senile hemangiomas. Although IVLBCL was known to demonstrate various CNS lesions, it is extremely rare to be manifested as a single lacunar infarction, and this case must be important for the differential diagnosis.
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http://dx.doi.org/10.5692/clinicalneurol.cn-001704DOI Listing
June 2022

[Recurrent autoimmune glial fibrillary acidic protein (GFAP) astrocytopathy with long cervical cord lesions: a case report].

Rinsho Shinkeigaku 2022 May 26;62(5):386-390. Epub 2022 Apr 26.

Department of Neurology, Nagaoka Red Cross Hospital.

A 61-year-old man who had suffered two episodes of generalized convulsion in a two-year period was admitted to our hospital because of progressive gait disturbance during the previous five months. Neurological examination revealed cognitive impairment, spasticity of the lower limbs, truncal ataxia, and dysautonomia including orthostatic hypotension, dysuria and hypohydrosis. Brain fluid-attenuated inversion recovery (FLAIR) MRI detected high-signal-intensity lesions in the periventricular white matter and centrum semiovale, with punctate gadolinium (Gd) enhancement. Spinal MRI detected swollen cervical long cord lesions extending from C2 to C6. Although methylprednisolone pulse treatment initially ameliorated the symptoms and MRI abnormal findings, clinical symptoms and MRI abnormalities including new cervical lateral column lesions reminiscent of those in autoimmune glial fibrillary acidic protein (GFAP) astrocytopathy (GFAP-A) relapsed two months later. At this point, anti-GFAPα antibody was detected in the cerebrospinal fluid. Although the clinical course of GFAP-A has been well reported, the present case showed a chronic refractory course.
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http://dx.doi.org/10.5692/clinicalneurol.cn-001713DOI Listing
May 2022

Orthostatic hemodynamics in the vertebral artery and blood pressure in patients with orthostatic dizziness/vertigo.

Auris Nasus Larynx 2022 Aug 18;49(4):593-598. Epub 2021 Dec 18.

Department of Otolaryngology-Head and Neck Surgery, Nara Medical University School of Medicine Address: 840 Shijo, Kashihara Nara, 634-8522, Japan.

Objectives: Orthostatic dizziness/vertigo (ODV) is a common symptom and is believed to occur due to the cerebral hypoperfusion caused by orthostatic hypotension (OH). However, the detailed mechanism underlying ODV onset is poorly understood. The vertebral artery (VA) mainly supplies blood to the central vestibular system; therefore, the orthostatic decrease of VA blood flow could possibly lead to ODV. This study investigated the orthostatic blood pressure and VA hemodynamics in ODV patients to elucidate the hemodynamic mechanism underlying ODV onset. Furthermore, the influence of orthostatic hypotension (OH) on VA hemodynamics was examined because OH is probably the most common cause of ODV.

Methods: This study included 181 patients with ODV and 73 control patients without ODV. All subjects underwent an active standing test to measure the extracranial Doppler (ECD) sonography spectrum of the VA and blood pressure (BP). VA blood flow velocity and BP were simultaneously measured for each patient in the supine static position and then in the upright standing positions following 3 min of standing. We investigated the orthostatic change in the average of flow velocity in bilateral VAs (VAFV) and BP for ODV patients compared with the control patients.

Result: VAFV in ODV patients was significantly reduced when standing up compared with the control patients. In the ODV patients, there was no difference in orthostatic decrease in VAFV between patients those with OH and without OH. However, the VAFV in the standing position was significantly lower in patients with OH than without OH. In cases with OH, the ODV patients exhibited a greater decrease in VAFV compared with the control patients, but this was not statistically significant. In the absence of OH, a significantly greater orthostatic decrease in VAFV was observed in ODV patients compared with the controls.

Conclusion: Our findings suggest that the orthostatic decrease of VA blood flow is deeply involved in the hemodynamic mechanism underlying ODV onset and is possibly associated with OH and other etiologies.
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http://dx.doi.org/10.1016/j.anl.2021.12.002DOI Listing
August 2022

[A case of neuromyelitis optica associated with pulmonary Mycobacterium avium complex disease].

Rinsho Shinkeigaku 2021 Sep 26;61(9):635-639. Epub 2021 Aug 26.

Department of Neurology, Nagaoka Red Cross Hospital.

A 62-year-old woman suffering from pulmonary Mycobacterium avium complex (MAC) disease was admitted to our hospital with fever, visual impairment, and lower limb weakness. MRI detected lesions in the optic chiasm and spinal cord extending the length of 6 vertebrae. The anti-aquaporin 4 (AQP4) antibody titer determined by ELISA was elevated to 8.3 IU/l. On the basis of these findings, the patient was diagnosed as having neuromyelitis optica (NMO), when chest CT also demonstrated exacerbation of pulmonary lesions. Methylprednisolone pulse therapy and double-filtered plasma exchange ameliorated the symptoms, and the EDSS score improved from 8.5 to 6.5. Six months later, visual impairment recurred, although ELISA showed that the anti-AQP4 antibody titer had become undetectable. Also, the CSF interleukin-6 (IL-6) level was elevated to 34.8 pg/ml. There have been few reports of NMO associated with pulmonary MAC disease. An increase of IL-6 is considered to exacerbate the clinical picture of NMO, whereas it may suppress progression of the pulmonary MAC disease. Exacerbation of the pulmonary MAC disease and the following internal counteraction with IL-6 may have resulted in a NMO relapse. The present patient was therefore administered eculizumab but not satralizumab, a humanized anti-IL-6 receptor antibody, for prevention of NMO recurrence.
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http://dx.doi.org/10.5692/clinicalneurol.cn-001614DOI Listing
September 2021

[Observing the time course of early long pyramidal tract Wallerian degeneration after acute ischemic stroke: a case report].

Rinsho Shinkeigaku 2021 Jul 18;61(7):477-481. Epub 2021 Jun 18.

Department of Neurology, Nagaoka Red Cross Hospital.

A 75-year-old man was found lying prostrate in a hot room in the middle of summer. On admission, he had high fever, dehydration, and multiple decubitus, in addition to right hemiparesis and total aphasia. Brain CT showed subacute ischemic stroke in the territory of left middle cerebral artery. Brain MRI diffusion-weighted imaging (DWI) 4 days after admission detected high signal intensity lesions in the left pyramidal tract from the midbrain cerebral peduncle to the lower pons, indicating early Wallerian degeneration. The lesions were found to extend to the contralateral pyramidal decussation by MRI DWI day 12, but they had disappeared on day 28. On the other hand, brain MRI FLAIR images detected the lesions clearly day 44. Also, diffusion tensor tractography detects fewer left cerebral pyramidal tracts. No previous reports have documented the time course of such long Wallerian degeneration. This case suggests that dehydration may promote the onset of early and long Wallerian degeneration.
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http://dx.doi.org/10.5692/clinicalneurol.cn-001588DOI Listing
July 2021

[Selenoprotein-related myopathy in a patient with old-age-onset type 2 respiratory failure: a case report].

Rinsho Shinkeigaku 2021 Apr 25;61(4):243-246. Epub 2021 Mar 25.

Department of Neurology, Nagaoka Red Cross Hospital.

A 71-year-old woman was admitted to our hospital with type2 respiratory failure. Her daily life activities had been normal, although she had noticed mild truncal weakness in her sixties. Her parents were consanguineous, and her sister had suffered similar symptoms. Although Pompe disease was suspected on the basis of the clinical course and CT findings of selective muscular atrophy in the paraspinal, thigh flexor and sartorius muscle, acid alpha-glucosidase activity was normal. The serum creatine kinase level was not elevated, and muscle biopsy showed no specific change. Genetic analysis revealed a novel homozygous variant c.227T>C (p.Phe76Ser) in the SELENON gene, and she was suspected to have selenoprotein-related myopathy, which is reported to develop in childhood. Selenoprotein-related myopathy should be considered as a differential diagnosis in aged patients presenting with respiratory failure of unknown origin.
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http://dx.doi.org/10.5692/clinicalneurol.cn-001544DOI Listing
April 2021

[Paraneoplastic anti-gamma aminobutyric acid (GABA) receptor antibody limbic encephalitis associated with small cell lung cancer presenting as new-onset status epilepticus: a case report].

Rinsho Shinkeigaku 2020 Dec 20;60(12):869-873. Epub 2020 Nov 20.

Department of Neurology, Nagaoka Red Cross Hospital.

An 84-year-old man was admitted to our hospital with new-onset refractory status epilepticus of unclear etiology. On the third day, diffusion-weighted brain MRI demonstrated lesions in the right medial temporal and parietal lobes. As a CSF sample showed pleocytosis, paraneoplastic limbic encephalitis (PLE) associated with small cell lung cancer (SCLC) was suspected. The patient was also positive for anti-gamma aminobutyric acid (GABA) receptor antibody in the CSF, which has recently been reported in elderly patients with SCLC-related PLE. Methylprednisolone pulse therapy ameliorated the symptoms. It is noteworthy that immune therapy often improves the symptoms of PLE with anti-GABA receptor antibody, even though radical therapy for the lung cancer may be difficult.
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http://dx.doi.org/10.5692/clinicalneurol.cn-001503DOI Listing
December 2020

Comparison of insulin degludec (IDeg)/insulin Aspart (IAsp) co-formulation therapy twice-daily with free combination of GLP-1 receptor agonist liraglutide plus insulin degludec in Tochigi: IDEAL Trial.

Int J Clin Pract 2021 Apr 9;75(4):e13734. Epub 2020 Nov 9.

Department of Endocrinology and Metabolism, Dokkyo Medical University, Mibu, Tochigi, Japan.

Aim: We compared the efficacy and safety of insulin degludec/insulin aspart co-formulation (IDegAsp) twice-daily to a free combination of basal insulin degludec and GLP-1 receptor agonist liraglutide (IDeg + Lira) once-daily for patients with inadequately controlled type 2 diabetes on insulin therapy and oral antidiabetic drugs.

Subjects And Methods: Eligible patients were randomly allocated at a 1:1 ratio to receive either the once-daily dual injection of IDeg + Lira (n = 24) or twice-daily single injection of IDegAsp (n = 28). The primary endpoints were as follows: HbA1c changes over 52 weeks of treatment and the percentage of participants achieving HbA1c < 7.0% at week 52.

Results: After 52 weeks, HbA1c decreased by 0.3% in the IDegAsp group and by 0.7% in the IDeg + Lira group. The HbA1c reduction was greater in the IDeg + Lira group than in the IDegAsp group. 19% of patients on IDegAsp versus 40% on IDeg + Lira achieved HbA1c < 7.0%. Pre-breakfast and pre-dinner blood glucose at 52 weeks were significantly lower in the IDeg + Lira group than in the IDegAsp group. The reduction in body mass index (BMI) was greater in the IDeg + Lira group than in the IDegAsp group throughout the study period. The confirmed hypoglycaemia rates were 1.32 and 0.69 per patient/year of exposure to IDegAsp and IDeg + Lira, respectively.

Conclusions: In patients with inadequately controlled type 2 diabetes on insulin therapy and oral antidiabetic drugs, treatment with the once-daily dual injection of IDeg + Lira compared with the twice-daily single injection of IDegAsp showed no significant difference in glycaemic control but statistically superior weight loss.
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http://dx.doi.org/10.1111/ijcp.13734DOI Listing
April 2021

[A case of thyrotoxic myopathy with generalized body muscular atrophy including the tongue muscle, lacking physical manifestations of Basedow disease].

Rinsho Shinkeigaku 2020 Oct 5;60(10):677-681. Epub 2020 Sep 5.

Department of Neurology, Nagaoka Red Cross Hospital.

We report a 74-year-old man with a 2-year history of proximal limb pain, body weight loss of 15 kg, and muscle weakness. Muscle atrophy was evident in the limbs and trunk, as well as the tongue. He was admitted to our hospital with suspected amyotrophic lateral sclerosis (ALS). Although he had no physical manifestations of Basedow disease such as palpitations, hyperhidrosis, hand tremor, exophthalmos, and an enlarged thyroid, he was diagnosed as having thyrotoxic myopathy as laboratory examinations indicated hyperthyroidism and positivity for TSH receptor antibody. The serum level of soluble IL-2 receptor was also elevated. Despite the severe muscle atrophy, the serum CK level was normal. A biopsy from the left quadriceps muscle revealed Type 1 fibers atrophy. Administration of anti-thyroid drugs normalized his thyroid function and the level of soluble IL-2 receptor, leading to improvement of the generalized muscle atrophy.
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http://dx.doi.org/10.5692/clinicalneurol.cn-001365DOI Listing
October 2020

PLA2G6 variants associated with the number of affected alleles in Parkinson's disease in Japan.

Neurobiol Aging 2021 01 13;97:147.e1-147.e9. Epub 2020 Jul 13.

Department of Neurology, National Hospital Organization Akita National Hospital, Yurihonjo, Akita, Japan.

This study aimed to evaluate genotype-phenotype correlations of Parkinson's disease (PD) patients with phospholipase A2 group V (PLA2G6) variants. We analyzed the DNA of 798 patients with PD, including 78 PD patients reported previously, and 336 in-house controls. We screened the exons and exon-intron boundaries of PLA2G6 using the Ion Torrent system and Sanger method. We identified 21 patients with 18 rare variants, such that 1, 9, and 11 patients were homozygous, heterozygous, and compound heterozygous, respectively, with respect to PLA2G6 variants. The allele frequency was approximately equal between patients with familial PD and those with sporadic PD. The PLA2G6 variants detected frequently were identified in the early-onset sporadic PD group. Patients who were homozygous for a variant showed more severe symptoms than those who were heterozygous for the variant. The most common variant was p.R635Q in our cohort, which was considered a risk variant for PD. Thus, the variants of PLA2G6 may play a role in familial PD and early-onset sporadic PD.
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http://dx.doi.org/10.1016/j.neurobiolaging.2020.07.004DOI Listing
January 2021

[A case of meningeal melanomatosis diagnosed by immunostaining of cerebrospinal fluid].

Rinsho Shinkeigaku 2020 Aug 7;60(8):565-568. Epub 2020 Jul 7.

Department of Neurology, Nagaoka Red Cross Hospital.

A 49-year-old woman was admitted to our hospital with suspected hypertensive encephalopathy. On the basis of MRI showing leptomeningeal enhancement and Class V cytology of the CSF, she was diagnosed as having leptomeningeal carcinomatosis. Although no primary site was detected, a few melanin granules were observed at the third CSF examination. The atypical cells in the CSF demonstrated immunoreactivity for HMB-45 and S-100, which are specific markers of malignant melanoma. There have been few reports of meningeal melanomatosis in Japan. This case illustrates that immunostaining is diagnostically useful in patients with leptomeningeal carcinomatosis from neoplasms with unknown primary sites.
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http://dx.doi.org/10.5692/clinicalneurol.60.cn-001436DOI Listing
August 2020

[A case of anti-titin antibody positive nivolumab-related necrotizing myopathy with myasthenia gravis].

Rinsho Shinkeigaku 2019 Jul 27;59(7):431-435. Epub 2019 Jun 27.

Department of Neurology, Nagaoka Red Cross Hospital.

A 53-year-old man suffering from squamous cell lung cancer presented with bilateral ptosis and bulbar palsy a month after initial treatment with the immune checkpoint inhibitor nivolumab. The symptoms showed worsening from midday, suggesting myasthenia gravis (MG), although anti-AChR antibody was negative. Although no muscle weakness was detected, the CK level was elevated to 5,255 IU/l, and MRI of the thigh revealed inflammation of the bilateral rectus femoris muscle. A muscle biopsy showed signs of necrotizing myopathy with expression of sarcolemmal HLA class I and accumulation of macrophages, CD4, CD8, and CD20-positive lymphocytes. Positivity for anti-titin antibody, one of the anti-striated muscle antibodies, was evident. The patient was diagnosed as having nivolumab-related necrotizing myopathy with myasthenia gravis, an immune-related adverse event (irAE). Treatment with prednisolone rapidly ameliorated the symptoms, and the serum CK level normalized. There have been several reports of nivolumab-related myositis with MG. On the basis of the muscle pathology and antibody data, we were able to clarify that necrotizing myopathy was related to the pathogenesis of this case.
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http://dx.doi.org/10.5692/clinicalneurol.cn-001270DOI Listing
July 2019

[A case of chordoma presenting as recurrent bacterial meningitis with cerebrospinal fluid leakage].

Rinsho Shinkeigaku 2019 May 26;59(5):264-267. Epub 2019 Apr 26.

Department of Neurology, Nagaoka Red Cross Hospital.

A 52-year-old man was admitted to our hospital because of two episodes of bacterial meningitis within a 6-month period. CSF examination showed neutrophilic pleocytosis with marked elevation of protein and hypoglycorrhachia, but the inflammatory reaction was mild and blood and CSF cultures were negative. At the time of the second admission, intermittent watery nasal discharge caused by CSF rhinorrhea was evident. CT and MR imaging revealed a tiny clival bone defect, and transnasal endoscopic repair was performed successfully. The pathological diagnosis was chordoma based on immunohistochemical staining for brachyury. Although chordoma presenting as recurrent bacterial meningitis occurs extremely rare, asking patients detailed questions about the CSF rhinorrhea must be essential for disclosing unclear infection sources.
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http://dx.doi.org/10.5692/clinicalneurol.cn-001272DOI Listing
May 2019

[A case of multiple cerebral hemorrhage caused by sudden increase of eosinophil in a patient with eosinophilic granulomatosis with polyangiitis].

Rinsho Shinkeigaku 2018 Sep 31;58(9):565-569. Epub 2018 Aug 31.

Department of Neurology, Nagaoka Red Cross Hospital.

A 42-year-old woman with bronchial asthma was admitted to our hospital due to sensory dominant mononeuritis multiplex lasting for more than 6 months. At that time, her eosinophil count was 761/μl and her sural nerve biopsy showed no findings suggestive of vasculitis. Four months later, she experienced sudden convulsions and right hemiparesis due to left lobular parietal subcortical hemorrhage, when her eosinophil count was elevated to 3,257/μl. Numerous microbleeds and small infarctions were also detected in the intracerebral areas of different regions with MRI. Eosinophilic granulomatosis with polyangiitis (EGPA) is a systemic necrotizing vasculitis of the small vessels, commonly affecting the peripheral nerves. Subarachnoid hemorrhage in patients with EGPA is extremely rare. Steep elevation of the eosinophil count may release certain cytokines, causing cerebral hemorrhage.
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http://dx.doi.org/10.5692/clinicalneurol.cn-001188DOI Listing
September 2018

Effect of insulin degludec versus insulin glargine on glycemic control and daily fasting blood glucose variability in insulin-naïve Japanese patients with type 2 diabetes: I'D GOT trial.

Diabetes Res Clin Pract 2017 Aug 15;130:237-243. Epub 2017 Jun 15.

Green Clinic, Mibu, Tochigi, Japan.

Aims: Insulin degludec (IDeg) is an ultra-long-acting insulin that has a smooth time/action profile over more than 42h. The present study compared the effects of IDeg and insulin glargine (IGlar) on HbA1c reduction and on within-subject day-to-day variability of fasting blood glucose (FBG) in insulin-naïve patients with type 2 diabetes.

Subjects And Methods: Eligible patients were randomly allocated at a 3:1 ratio to receive once-daily IDeg (n=31) or IGlar (n=12). Both basal insulins were administered before breakfast and titrated to achieve a target FBG <110mg/dl. The primary endpoints were the change in HbA1c from baseline to 24weeks of treatment, as well as the standard deviation (SD) and coefficient of variation (CV) of FBG from 8 to 12weeks and from 20 to 24weeks. Secondary endpoints included the QOL evaluated by the Diabetes Therapy-Related QOL questionnaire.

Results: After 24weeks, HbA1c was decreased by 1.6% in the IDeg group and 1.7% in the IGlar at the same insulin dosage. At 24weeks, FBG was significantly lower in the IDeg group than in the IGlar group and the CV of FBG was significantly smaller in the IDeg group. The frequency of total and severe hypoglycemic episodes did not differ between the groups. In the IDeg group, QOL showed significant improvement regarding anxiety and dissatisfaction with treatment.

Conclusions: Treatment with IDeg or IGlar achieved similar improvement in glycemic control in insulin-naïve patients with type 2 diabetes. The day-to-day variation of FBG was smaller in patients receiving IDeg.
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http://dx.doi.org/10.1016/j.diabres.2017.06.007DOI Listing
August 2017

Herpes simplex encephalitis presenting as stroke-like symptoms with atypical MRI findings and lacking cerebrospinal fluid pleocytosis.

Rinsho Shinkeigaku 2017 07 21;57(7):387-390. Epub 2017 Jul 21.

Department of Neurology, Nagaoka Red Cross Hospital.

A 73-year-old woman presented with sudden onset of right hemiparesis and was diagnosed as having cerebral infarction on the basis of diffusion-weighted brain MRI, which demonstrated lesions in the left parietal cortex. On the 3rd day, the patient developed right upper limb myoclonus, aphasia, and disturbance of consciousness with high fever. On the 6th day, she was transferred to our hospital with suspected viral encephalitis, and treatment with acyclovir was started. By the 6th day, the lesions detected by MRI had expanded to the gyrus cinguli, insula and thalamus, but not to the temporal lobe. At that time, the CSF cell count was 8/μl, and this later increased to 17/μl by the 13th day. Although herpes simplex virus DNA was detected in the CSF on the 6th day, there was no evidence of CSF pleocytosis or temporal lobe abnormalities demonstrable by brain MRI throughout the whole follow-up period. This was very atypical case of herpes simplex encephalitis characterized by a stroke-like episode, atypical MRI findings, and absence of cerebrospinal fluid pleocytosis. It is important to be mindful that herpes simplex encephalitis (HSE) can have an atypical presentation, and that sufficient acyclovir treatment should be initiated until HSE can be ruled out.
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http://dx.doi.org/10.5692/clinicalneurol.cn-001033DOI Listing
July 2017

[A case of conus medullaris infarction expanding to the vertebral bodies, major psoas and erector spinae muscles].

Rinsho Shinkeigaku 2015 11;55(9):661-4. Epub 2015 Jul 11.

Department of Neurology, Nagaoka Red Cross Hospital.

A 77-year-old woman presented with conus medullaris and cauda equina syndrome following a sudden pain in the bilateral lower abdomen and right buttock. Lumbar magnetic resonance imaging (MRI) showed not only a conus medullaris lesion, but also several lesions in the vertebral bodies (L1, L2), right major psoas muscle, right multifidus muscle and bilateral erector spinae muscles. As these areas receive blood supply from each branch of the same segmental artery, we considered all of the lesions as infarctions that were a result of a single parent vessel occlusion. It is known that a vertebral body lesion can be accompanied by a spinal cord infarction, but in combination with infarction of a muscle has not been reported. This is the first report of a concomitant spinal cord and muscle infarction revealed by MRI. It is noteworthy that a spinal cord infarction could expand not only to neighboring vertebral bodies, but also to muscles.
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http://dx.doi.org/10.5692/clinicalneurol.cn-000729DOI Listing
April 2016

[Transformation from chronic subdural hematoma into subdural empyema following cat bites: a case report].

Rinsho Shinkeigaku 2015 11;55(9):657-60. Epub 2015 Jul 11.

Department of Neurology, Nagaoka Red Cross Hospital.

A 69-year-old man developed motor aphasia and right hemiparesis with severe headache, during the treatment of cellulitis and sepsis due to cat bites. Brain CT showed a low density, crescent-shaped lesion in the left subdural space, which was hypointense on brain diffusion-weighted imaging (DWI). One week later, when his neurological symptoms had worsened, the signal of the subdural lesion had changed to hyperintense on DWI. The lesion was capsule-shaped when enhanced by Gadolinium. The signal changes on DWI of the lesion indicated the existing hematoma had changed to an empyema, or so-called infected subdural hematoma, due to a hematogenous bacterial infection. Pasteurella multocida, a resident microbe in the oral cavity of cats, could be the responsible pathogen in this case. The patient recovered completely after treatment with intravenous high dose antibiotics. This is an important case report describing the transformation from a chronic subdural hematoma into a subdural empyema by DWI.
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http://dx.doi.org/10.5692/clinicalneurol.cn-000725DOI Listing
April 2016

[A case of Behçet disease developing recurrent ischemic stroke with fever and scrotal ulcers].

Rinsho Shinkeigaku 2015 ;55(6):428-31

Department of Neurology, Nagaoka Red Cross Hospital.

A 30-year-old man, who was diagnosed with Behçet disease at 10 years of age, was hospitalized because of transient right hemiparesis after presenting with high fever and scrotal ulcers. Brain MRI revealed ischemic lesions in the area supplied by the anterior cerebral arteries. Analysis of cerebrospinal fluid (CSF) showed pleocytosis and a high interleukin-6 (IL-6) concentration (668 pg/ml). The patient was diagnosed with acute ischemic stroke associated with exacerbation of Behçet disease. After initiation of corticosteroid therapy, his clinical symptoms improved, and the CSF IL-6 concentration decreased. One year later, the patient developed high fever and scrotal ulcers after the onset of transient left upper limb plegia. Brain MRI showed an acute ischemic lesion in the right putamen, and CSF analysis showed an elevated IL-6 concentration (287 pg/ml). Brain CT angiography revealed stenosis of the left anterior cerebral artery and occlusion of the right anterior cerebral artery, which had been well visualized one year previously. Involvement of the intracranial cerebral arteries in Behçet disease is extremely rare. To the best of our knowledge, this is the first case report of a patient with recurrent symptomatic ischemic stroke associated with high fever and scrotal ulcers, which suggests exacerbation of Behçet disease.
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http://dx.doi.org/10.5692/clinicalneurol.cn-000682DOI Listing
March 2016

[A case of progressive encephalomyelitis with rigidity and myoclonus associated with anti-GAD, anti-glycine receptor and anti-GM1 antibodies].

Rinsho Shinkeigaku 2015 ;55(2):111-4

Department of Neurology, Nagaoka Red Cross Hospital.

A 62-year-old woman with one-year history of type 1 diabetes mellitus was admitted to our hospital with progressive weakness in the lower extremities and urinary dysfunction following high fever. On admission, she had rigidity and myoclonus in the upper extremities with sensory ataxia. Cerebrospinal fluid examination revealed mild pleocytosis and oligoclonal band. Glutamic acid decarboxylase (GAD) antibodies were detected at high titer in serum, but antibodies to glycine receptor (GlyR), thyroid peroxidase, mitochondrial M2, and GM1 were also detected. She was diagnosed with progressive encephalomyelitis with rigidity and myoclonus (PERM), which probably developed on the basis of polyglandular autoimmune syndromes. The clinical symptoms began to improve after initiation of intravenous high-dose methylprednisolone. Muscle weakness might be related to GM1 antibodies. This is the first report of PERM, in which GM1 antibodies were detected with GAD and GlyR antibodies.
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http://dx.doi.org/10.5692/clinicalneurol.55.111DOI Listing
September 2016

The prevalence and characteristics of metabolic syndrome in patients with vertigo.

PLoS One 2013 3;8(12):e80176. Epub 2013 Dec 3.

Department of Otolaryngology-Head and Neck Surgery, Nara Medical University School of Medicine, Kashihara, Nara, Japan.

Objectives/hypothesis: Metabolic syndrome (MetS) is a condition that increases the risk of coronary artery disease and cerebral infarction. We determined the prevalence of MetS in vertigo patients and clinically investigated the association between MetS and vertigo.

Study Design: Case-control study.

Methods: The subjects were 333 patients, including 107 males and 226 females, who presented with vertigo as a primary symptom. MetS was diagnosed according to the International Diabetes Federation definition, which is based on waist circumference, blood serum levels, and blood pressure.

Results: MetS was detected in 53 (15.9%) of 333 vertigo patients, including 24 males (22.4%) and 29 females (12.8%); i.e., the frequency of MetS was significantly higher among the male patients than the female patients. The overall prevalence of MetS (15.9%) among vertigo patients did not differ from that observed among general adults in previous Japanese surveillance studies; however, MetS was significantly more common among the vertigo patients in males than general adult males. The prevalence of MetS was also examined in five types of vertigo, Concomitant MetS was noted in many males with vertebrobasilar insufficiency (VBI) and isolated vertigo of unknown etiology.

Conclusion: It was suggested that MetS is involved in the development of vertigo in males. MetS might be a risk factor for vascular vertigo such as VBI in males. The high frequency of MetS among males with vertigo of unknown etiology suggested that the pathogenesis of metabolic syndrome is involved in this type of isolated vertigo.
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http://journals.plos.org/plosone/article?id=10.1371/journal.pone.0080176PLOS
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC3848922PMC
August 2014

New treatment strategy for cupulolithiasis associated with benign paroxysmal positional vertigo of the lateral canal: the head-tilt hopping exercise.

Eur Arch Otorhinolaryngol 2014 Dec;271(12):3155-60

This study was performed to determine whether a novel treatment was effective against cupulolithiasis associated with benign paroxysmal positional vertigo (BPPV) of the lateral semicircular canal, which is characterized by apogeotropic direction-changing nystagmus. We herein describe our head-tilt hopping (HtH) exercise, which is designed to release otoconial debris strongly adhered to the cupula. The subjects were trained to hop while tilting their heads laterally. They completed 3 to 5 exercise sessions per day over a 4-week period. Each session ended with a 20-hop trial. The HtH exercises were performed by 27 patients with intractable lateral canal BPPV who exhibited positional vertigo and persistent nystagmus beating toward the uppermost ear for more than 4 weeks, despite performing therapeutic head shaking in the horizontal plane maneuver. All the patients were subjected to the supine roll test before and immediately after the first trial as well as after 1 and 4 weeks of the program to evaluate the effect of the treatment on their apogeotropic nystagmus. Nystagmus of 9 (33.3 %) patients disappeared immediately after the first training session. After 1 and 4 weeks of the training, the number of patients that had experienced either of these improvements had increased to 15 (55.6 %) and 19 (70.4 %) subjects, respectively. These results suggest that HtH exercises aimed at releasing otoconial debris from the cupula are feasible as a new therapy for cupulolithiasis associated with intractable lateral canal BPPV. However, further studies for comparison with control are required to confirm these preliminary results.
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http://dx.doi.org/10.1007/s00405-013-2808-2DOI Listing
December 2014

Predictors of outcome in hypoglycemic encephalopathy.

Diabetes Res Clin Pract 2013 Aug 29;101(2):159-63. Epub 2013 Jun 29.

Department of Neurology, Brain Research Institute, Niigata University, Japan.

Aims: The aim of this study was to investigate factors predicting poor prognosis in patients with hypoglycemic encephalopathy.

Methods: We retrospectively analyzed data on 165 consecutive patients with hypoglycemic encephalopathy. We evaluated their outcome 1 week after hypoglycemia onset using the Glasgow outcome scale (GOS) and compared the clinical features of patients with good outcomes (GOS = 5) and poor outcomes (GOS ≤ 4).

Results: The poor-outcome group included 38 patients (23%). The initial blood glucose level in the poor-outcome group was lower than that in the good-outcome group (p = 0.002). The duration of hypoglycemia in the poor-outcome group was longer than that in the good-outcome group (p < 0.001). Body temperature during hypoglycemia in the poor-outcome group was higher than that in the good-outcome group (p < 0.001). Furthermore, lactic acid level in the poor-outcome group was lower than in the good-outcome group (p = 0.032). There was no significant difference in the frequency of posttreatment hyperglycemia between the good-outcome and poor-outcome groups (p = 0.984).

Conclusion: Profound and prolonged hypoglycemia, normal or higher body temperature, and a low lactic acid level during hypoglycemia may be predictors of a poor outcome in patients with hypoglycemic encephalopathy.
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http://dx.doi.org/10.1016/j.diabres.2013.05.007DOI Listing
August 2013

Osteoporosis as a risk factor for the recurrence of benign paroxysmal positional vertigo.

Laryngoscope 2013 Nov 8;123(11):2813-6. Epub 2013 Apr 8.

Department of Otolaryngology-Head and Neck Surgery, Nara Medical University School of Medicine, Nara, Japan; and the.

Objectives/hypothesis: Benign paroxysmal positional vertigo (BPPV) frequently occurs in females over 50 years old, suggesting that a postmenopausal decrease in estrogen secretion might be involved in its onset. An estrogen deficiency is generally known to cause osteoporosis through a reduction in bone mass. This study was designed to investigate a clinical association between idiopathic BPPV and osteoporosis.

Study Design: Case-control study.

Methods: We measured the bone mineral density (BMD) at the lumbar vertebrae in 61 patients with idiopathic BPPV who were postmenopausal women over 50 years old using dual-energy x-ray absorptiometry. After being treated with the canalith repositioning maneuver, the patients were followed up for at least 1 year.

Results: Our results showed that the incidence of osteoporosis in patients with BPPV was 26.2%, which was similar to those observed in epidemiological surveys conducted in Japan. However, we found that in BPPV patients with osteoporosis, the incidence of recurrence was 56.3%, which was significantly higher than that observed in patients with normal bone mineral density (16.1%). Furthermore, the frequency of BPPV recurrence increased as BMD decreased.

Conclusions: These results suggest that osteoporosis is a risk factor for BPPV recurrence. The prognosis of BPPV might be clinically predicted by BMD reduction.
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http://dx.doi.org/10.1002/lary.24099DOI Listing
November 2013

[Neurolymphomatosis presenting as bilateral tongue atrophy: a case report].

Rinsho Shinkeigaku 2012 ;52(8):589-91

Department of Neurology, Nagaoka Red Cross Hospital.

A 62-year-old woman had progressive dysarthria for 2 months and was suspected of having amyotrophic lateral sclerosis because of the presentation of bilateral tongue atrophy and fasciculation. Brain magnetic resonance imaging (MRI) showed enlargement of the left hypoglossal nerve, and whole-body gallium scintigraphy showed abnormal uptake in the left pelvic cavity and left thigh. On the basis of the findings of biopsy of the mass lesion in the left thigh, she was diagnosed with diffuse large B-cell lymphoma. After chemotherapy for diffuse large B-cell lymphoma, the tongue atrophy improved. The patient subsequently developed left oculomotor nerve palsy, weakness of the right arm, and weakness of the right leg. The cause of these symptoms was thought to be neurolymphomatosis on the basis of the typical MRI findings observed. We report a rare case of neurolymphomatosis presenting as bilateral tongue atrophy, mimicking amyotrophic lateral sclerosis.
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http://dx.doi.org/10.5692/clinicalneurol.52.589DOI Listing
December 2013

[A case of CADASIL without characteristic anterior temporal pole lesion diagnosed by skin biopsy].

Rinsho Shinkeigaku 2011 Oct;51(10):770-3

Department of Neurology, Nagaoka Red Cross Hospital.

A 40 year-old man with migraine presented cerebral ischemic attacks several times in one year. He had no risk factors for cerebrovascular disease including hypertension, but had strong family history suggesting autosomal dominant inheritance. A brain MRI on T(2) weighted and FLAIR images revealed patchy and confluent hyper intensity areas in the subcortical white matters and bilateral external capsules, while no anterior temporal pole lesions characteristic of CADASIL (cerebral autosomal dominant arteriopathy with subcortical infarcts and leukoencephalopathy) were detected. His skin biopsy demonstrated granular osmiophilic materials (GOM) on the basement membrane of the vascular smooth muscle cells in dermis as shown by an electron microscope. The following mutational analysis of the Notch3 gene disclosed a missense mutation of p.Arg133Cys in exon 3. Molecular diagnosis of CADASIL may be time consuming because Notch3 is a huge gene and mutations may occur at multiple sites. GOM on skin biopsy is diagnostic especially in cases where anterior temporal pole involvement on MRI is negative.
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http://dx.doi.org/10.5692/clinicalneurol.51.770DOI Listing
October 2011

[A case of inflammatory myopathy with widely skin rash following use of supplements containing Spirulina].

Rinsho Shinkeigaku 2011 May;51(5):330-3

Department of Neurology, Nagaoka Red Cross Hospital.

A 49-year old woman noticed her skin rash several days after taking supplements containing Spirulina, a planktonic blue-green alga. Her skin rash was spreading over large parts of her body, even after stop ingestion two months later. Five months later, she developed muscle weakness of neck flexor and left proximal upper extremity. On admission, creatine kinase (CK) was elevated to 1,268 IU/ml in the serum. A muscle specimen revealed many necrotizing muscle fibers and the infiltration of mononuclear cells in the peri- and endomysium including a lot of eosinophils. Immunohistochemical staining showed the infiltration of CD4 positive cells in the peri- and endomysium and that of CD20 positive B cells in the perivascular regions. She was diagnosed as having inflammatory myopathy with widely skin rash. Therapy with administration of prednisolone and cyclophosphamide followed by methyl-prednisolone pulse improved her clinical symptoms. There is a similar report describing a case of dermatomyositis after ingestion of Spirulina, which is known to have immune-stimulating property such as accelerating tumor necrosis factor (TNF)-alpha production. Also, TNF-alpha single nucleotide polymorphisms (TNF-308A) was demonstrated to have strong association with onset of myositis in Caucasians. The use of Spirulina could result in inflammatory myopathy under some specific conditions.
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http://dx.doi.org/10.5692/clinicalneurol.51.330DOI Listing
May 2011

Vertebral basilar system dolichoectasia with marked infiltration of IgG4-containing plasma cells: a manifestation of IgG4-related disease?

Neuropathology 2012 Feb 26;32(1):100-4. Epub 2011 May 26.

Department of Pathology, Brain Research Institute, University of Niigata, Japan.

We report the histopathological features of vertebral basilar system dolichoectasia (VBD) in a 68-year-old man who died as a result of accompanying infarction of the medulla oblongata on day 6 of admission. During hospitalization, the patient was also found to have an elevated serum IgG level and tumors of the renal pelvis. A possible clinical diagnosis of VBD associated with IgG4-related disease was considered postmortem. Autopsy examination, limited to the intracranial tissues, revealed marked infiltration of IgG4-containing plasma cells in the adventitia and media of the vertebral and basilar arteries. Multiple fibrous nodules forming pseudotumors were also evident on the outer surface of the affected arteries. These histological features were very similar to those of arteriopathy, such as inflammatory aortic aneurysm, which has been described in patients with IgG4-related disease, suggesting that autoimmune mechanisms, known to be involved in the pathogenesis of visceral lesions in the disease, also played a role in the etiology of VBD in the present patient. In conclusion, we consider that the present case may represent VBD as a manifestation of IgG4-related disease.
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http://dx.doi.org/10.1111/j.1440-1789.2011.01227.xDOI Listing
February 2012

[Clinical analysis of metabolic syndrome in vertiginous diseases].

Nihon Jibiinkoka Gakkai Kaiho 2011 Jan;114(1):24-9

Department of Otolaryngology-Head and Neck Surgery, Nara Medical University School of Medicine, Kashihara.

To explore the relationship between metabolic syndrome and vertigo, we measured waist circumference, plasma glucose, triglycerides and blood pressure in 333 subjects aged 20-79 years with vertigo. We found overall metabolic syndrome prevalence defined by Japanese diagnostic criteria to be 13.2%, similar to that in other national surveys by the Japanese Ministry of Health, Labour and Welfare. The 6-fold higher prevalence in men over women exceeded that of other reports, however. The highest frequency was in vertebrobasilar insufficiency (VBI) disorders, suggesting that conditions such as VBI in men with vertigo could involve metabolic syndrome as a risk factor for vertigo incidence.
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http://dx.doi.org/10.3950/jibiinkoka.114.24DOI Listing
January 2011
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