Pub Date : 2017-01-28DOI: 10.5692/CLINICALNEUROL.CN-000891
Masayoshi Yamamoto, Manabu Inoue, Naoko Tachibana, Koji Tsuzaki, Yoko Shibata, T. Hamano
The patient was a 36-year-old man. His initial symptom was bilateral thigh and calf pain. When he developed ulcerative colitis in the following year, he also noticed wasting of the calf muscles. The clinical feature is similar to gastrocnemius myalgia syndrome, although the left upper limb was also involved. A high-intensity lesion in the left calf and soleus muscles was observed on MRI, which was lead to the diagnosis of non-granulomatous myositis with infiltration of CD68-positive cells based on muscle biopsy. After steroids were administered, his pain subsided. Evaluation with needle EMG, MRI, and muscle biopsy is important when muscle pain accompanies inflammatory bowel disease.
{"title":"潰瘍性大腸炎に合併しgastrocnemius myalgia syndrome様の症状を呈した非肉芽腫性筋炎の1例","authors":"Masayoshi Yamamoto, Manabu Inoue, Naoko Tachibana, Koji Tsuzaki, Yoko Shibata, T. Hamano","doi":"10.5692/CLINICALNEUROL.CN-000891","DOIUrl":"https://doi.org/10.5692/CLINICALNEUROL.CN-000891","url":null,"abstract":"The patient was a 36-year-old man. His initial symptom was bilateral thigh and calf pain. When he developed ulcerative colitis in the following year, he also noticed wasting of the calf muscles. The clinical feature is similar to gastrocnemius myalgia syndrome, although the left upper limb was also involved. A high-intensity lesion in the left calf and soleus muscles was observed on MRI, which was lead to the diagnosis of non-granulomatous myositis with infiltration of CD68-positive cells based on muscle biopsy. After steroids were administered, his pain subsided. Evaluation with needle EMG, MRI, and muscle biopsy is important when muscle pain accompanies inflammatory bowel disease.","PeriodicalId":371642,"journal":{"name":"Rinshō shinkeigaku Clinical neurology","volume":"24 1","pages":"0"},"PeriodicalIF":0.0,"publicationDate":"2017-01-28","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"133494100","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
A 79 year-old female was admitted to our hospital because of high serum creatine kinase level together with proximal muscle weakness and pain on grasping. MRI revealed inflammatory changes in femoral muscles on both sides. Muscle biopsy showed size irregularity of muscle cells, and necrosis and regeneration of fibers. Study of antibodies was also consistent with the diagnostic criteria of anti-signal recognition particle (anti-SRP) antibody-positive myopathy. On admission, the patient required pericardiocentesis for the management of exudative pericarditis. Accompanying the aggravation of myositis, negative T wave in precordial leads on ECG, ventricular extrasystoles and non-sustained ventricular tachycardia were observed. These abnormalities were resolved with the improvement of myositis by immunosuppressive treatment. These observations suggest that the myopericarditis was associated with anti-SRP antibody-positive myopathy.
{"title":"心膜心筋炎の合併が疑われた抗signal recognition particle（SRP）抗体陽性ミオパチーの1例","authors":"Mariko Tanaka, Naoki Gamou, Hirohiko Shizukawa, Emiko Tsuda, Shun Shimohama","doi":"10.5692/CLINICALNEUROL.CN-000916","DOIUrl":"https://doi.org/10.5692/CLINICALNEUROL.CN-000916","url":null,"abstract":"A 79 year-old female was admitted to our hospital because of high serum creatine kinase level together with proximal muscle weakness and pain on grasping. MRI revealed inflammatory changes in femoral muscles on both sides. Muscle biopsy showed size irregularity of muscle cells, and necrosis and regeneration of fibers. Study of antibodies was also consistent with the diagnostic criteria of anti-signal recognition particle (anti-SRP) antibody-positive myopathy. On admission, the patient required pericardiocentesis for the management of exudative pericarditis. Accompanying the aggravation of myositis, negative T wave in precordial leads on ECG, ventricular extrasystoles and non-sustained ventricular tachycardia were observed. These abnormalities were resolved with the improvement of myositis by immunosuppressive treatment. These observations suggest that the myopericarditis was associated with anti-SRP antibody-positive myopathy.","PeriodicalId":371642,"journal":{"name":"Rinshō shinkeigaku Clinical neurology","volume":"47 1","pages":"0"},"PeriodicalIF":0.0,"publicationDate":"2016-11-25","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"116864022","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Pub Date : 2016-10-21DOI: 10.5692/CLINICALNEUROL.CN-000925
Jun Takei, Ran Takei, Satoshi Nozuma, Kei Nakahara, O. Watanabe, Hiroshi Takashima
: A 40-year-old man presented with a series of generalized tonic-clonic seizures after febrile illness. He developed status epilepticus and required mechanical ventilation with anesthetics. Steroid pulse, intravenous immunoglobulin, and immunoadsorption therapy were administrated, and the status epilepticus improved; however, drug-resistant seizures remained. Despite the use of several antiepileptic drugs, seizures frequently occurred. Additional administration of potassium bromide resulted in significant suppression of seizures. Potassium bromide is regarded as an effective medication for pediatric refractory epilepsy after encephalitis. The present case is considered to be new-onset refractory status epilepticus (NORSE) syndrome based on clinical features, and potassium bromide could be effective in treating adult refractory epilepsy, such as NORSE syndrome.
{"title":"難治性てんかんに臭化カリウムが奏効したnew-onset refractory status epilepticus syndromeの1例","authors":"Jun Takei, Ran Takei, Satoshi Nozuma, Kei Nakahara, O. Watanabe, Hiroshi Takashima","doi":"10.5692/CLINICALNEUROL.CN-000925","DOIUrl":"https://doi.org/10.5692/CLINICALNEUROL.CN-000925","url":null,"abstract":": A 40-year-old man presented with a series of generalized tonic-clonic seizures after febrile illness. He developed status epilepticus and required mechanical ventilation with anesthetics. Steroid pulse, intravenous immunoglobulin, and immunoadsorption therapy were administrated, and the status epilepticus improved; however, drug-resistant seizures remained. Despite the use of several antiepileptic drugs, seizures frequently occurred. Additional administration of potassium bromide resulted in significant suppression of seizures. Potassium bromide is regarded as an effective medication for pediatric refractory epilepsy after encephalitis. The present case is considered to be new-onset refractory status epilepticus (NORSE) syndrome based on clinical features, and potassium bromide could be effective in treating adult refractory epilepsy, such as NORSE syndrome.","PeriodicalId":371642,"journal":{"name":"Rinshō shinkeigaku Clinical neurology","volume":"34 1","pages":"0"},"PeriodicalIF":0.0,"publicationDate":"2016-10-21","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"123806972","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Pub Date : 2016-10-21DOI: 10.5692/CLINICALNEUROL.CN-000927
規絵 伊藤, 秀敏 竹井, 進 千葉, 菊郎 福島
: We reported recently that during a memory-based smooth-pursuit task, most Parkinson's disease (PD) patients exhibited normal cue-information memory but impaired smooth-pursuit preparation and execution. A minority of PD patients had abnormal cue-information memory or difficulty in understanding the task. To further examine differences between these two groups, we assigned an anti-saccade task and compared correct rates with various neuropsychological and motor symptom evaluations. The anti-saccade task requires voluntary saccades in the opposite direction to a visual stimulus, and patients with frontal cortical impairments are known to exhibit reflexive saccades (errors). We classified PD patients into 2 groups: one with normal cue-information memory during memory-based smooth-pursuit (n = 14), and the other with abnormal cue-information memory or with difficulty in understanding the memory task (n = 6). The two groups had significantly different anti-saccade correct rates and frontal assessment battery (FAB) scores (P < 0.01). Anti-saccade correct rates of individual patients (n = 20) correlated significantly with FAB scores (P < 0.01) but not with age, Hoehn-Yahr stage, unified PD rating scale (UPDRS) part III or mini-mental state examination (MMSE) scores. Among FAB subtests, significant correlation was obtained only with motor programming scores. These results suggest that performance of memory-based smooth-pursuit and/or anti-saccades depend on frontal cortical function or dysfunction.
{"title":"Parkinson’s disease（PD）における前頭葉機能不全：記憶追跡課題，anti-saccade課題と運動・高次脳機能評価の比較","authors":"規絵 伊藤, 秀敏 竹井, 進 千葉, 菊郎 福島","doi":"10.5692/CLINICALNEUROL.CN-000927","DOIUrl":"https://doi.org/10.5692/CLINICALNEUROL.CN-000927","url":null,"abstract":": We reported recently that during a memory-based smooth-pursuit task, most Parkinson's disease (PD) patients exhibited normal cue-information memory but impaired smooth-pursuit preparation and execution. A minority of PD patients had abnormal cue-information memory or difficulty in understanding the task. To further examine differences between these two groups, we assigned an anti-saccade task and compared correct rates with various neuropsychological and motor symptom evaluations. The anti-saccade task requires voluntary saccades in the opposite direction to a visual stimulus, and patients with frontal cortical impairments are known to exhibit reflexive saccades (errors). We classified PD patients into 2 groups: one with normal cue-information memory during memory-based smooth-pursuit (n = 14), and the other with abnormal cue-information memory or with difficulty in understanding the memory task (n = 6). The two groups had significantly different anti-saccade correct rates and frontal assessment battery (FAB) scores (P < 0.01). Anti-saccade correct rates of individual patients (n = 20) correlated significantly with FAB scores (P < 0.01) but not with age, Hoehn-Yahr stage, unified PD rating scale (UPDRS) part III or mini-mental state examination (MMSE) scores. Among FAB subtests, significant correlation was obtained only with motor programming scores. These results suggest that performance of memory-based smooth-pursuit and/or anti-saccades depend on frontal cortical function or dysfunction.","PeriodicalId":371642,"journal":{"name":"Rinshō shinkeigaku Clinical neurology","volume":"1 1","pages":"0"},"PeriodicalIF":0.0,"publicationDate":"2016-10-21","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"127343596","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Pub Date : 2016-10-21DOI: 10.5692/CLINICALNEUROL.CN-000930
貴也 北野, 允 木下, 宏樹 島津, 博彰 伏見, 謙一 大森, 敬憲 狭間
A 87-year-old female presented with subacute progression of cognitive decline. Fluid-attenuated inversion recovery images of brain MRI showed multifocal high-intensity lesions. Thoracic CT image revealed the presence of thymoma, and serum autoantibody screening showed positivity for anti-gamma aminobutyric acid (GABA)A receptor antibody. Histopathological analysis confirmed type B3 thymoma after thymectomy. The patient received both plasmapheresis and intravenous methylprednisolone therapy, and showed remarkable amelioration of clinical symptoms and MRI abnormal high intensity. However, after 2 month from the clinical recovery, the patient showed recurrence of brain lesions and intravenous methylprednisolone monotherapy was performed. Continuation of oral steroid therapy was required to maintain the quienscent state of inflammation within the central nervous system. Anti-GABAA receptor antibody is a recently discovered novel autoantibody associated with autoimmue encephalitis. Due to the limited number of literature reported, clinical course and therapeutic response of GABAA receptor antibody encephalitis remains elusive. Here we reported a rare case of GABAA receptor antibody encephalitis with type B3 thymoma. Clinical, radiological and therapeutic courses described in our report highlight the importance of immunotherapy for treatment of the disease.
{"title":"Type B3胸腺腫を合併した抗gamma aminobutyric acid （GABA）A受容体抗体陽性再発性脳炎の1例","authors":"貴也 北野, 允 木下, 宏樹 島津, 博彰 伏見, 謙一 大森, 敬憲 狭間","doi":"10.5692/CLINICALNEUROL.CN-000930","DOIUrl":"https://doi.org/10.5692/CLINICALNEUROL.CN-000930","url":null,"abstract":"A 87-year-old female presented with subacute progression of cognitive decline. Fluid-attenuated inversion recovery images of brain MRI showed multifocal high-intensity lesions. Thoracic CT image revealed the presence of thymoma, and serum autoantibody screening showed positivity for anti-gamma aminobutyric acid (GABA)A receptor antibody. Histopathological analysis confirmed type B3 thymoma after thymectomy. The patient received both plasmapheresis and intravenous methylprednisolone therapy, and showed remarkable amelioration of clinical symptoms and MRI abnormal high intensity. However, after 2 month from the clinical recovery, the patient showed recurrence of brain lesions and intravenous methylprednisolone monotherapy was performed. Continuation of oral steroid therapy was required to maintain the quienscent state of inflammation within the central nervous system. Anti-GABAA receptor antibody is a recently discovered novel autoantibody associated with autoimmue encephalitis. Due to the limited number of literature reported, clinical course and therapeutic response of GABAA receptor antibody encephalitis remains elusive. Here we reported a rare case of GABAA receptor antibody encephalitis with type B3 thymoma. Clinical, radiological and therapeutic courses described in our report highlight the importance of immunotherapy for treatment of the disease.","PeriodicalId":371642,"journal":{"name":"Rinshō shinkeigaku Clinical neurology","volume":"23 1","pages":"0"},"PeriodicalIF":0.0,"publicationDate":"2016-10-21","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"117003934","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Pub Date : 2016-01-21DOI: 10.5692/CLINICALNEUROL.CN-000771
Kenju Hara, Keigo Onda, Haruka Ouchi, Ken Shibano, Hideaki Ishiguro
: A 56-year-old man, who presented with 6 years history of difficulty in walking, was diagnosed as having vascular parkinsonism on the basis of the clinical findings of parkinsonism, pyramidal sign and the brain MRI findings of multiple lacunar infarction. Although he did not have hypertension, he had hyperhomocysteinemia and homozygous methylenetetrahydrofolate reductase (MTHFR) gene variant (C677T) as risk factors for ischemic stroke. Recent studies have shown that hyperhomocysteinemia and MTHFR gene variant are associated with small-vessel disease, suggesting that these risk factors may underlie vascular parkinsonism, particularly in patients lacking hypertension and in those with a relatively younger age at onset of this disease.
{"title":"高ホモシステイン血症とmethylenetetrahydrofolate reductase遺伝子多型（C677T）をみとめた脳血管性パーキンソニズムの1例","authors":"Kenju Hara, Keigo Onda, Haruka Ouchi, Ken Shibano, Hideaki Ishiguro","doi":"10.5692/CLINICALNEUROL.CN-000771","DOIUrl":"https://doi.org/10.5692/CLINICALNEUROL.CN-000771","url":null,"abstract":": A 56-year-old man, who presented with 6 years history of difficulty in walking, was diagnosed as having vascular parkinsonism on the basis of the clinical findings of parkinsonism, pyramidal sign and the brain MRI findings of multiple lacunar infarction. Although he did not have hypertension, he had hyperhomocysteinemia and homozygous methylenetetrahydrofolate reductase (MTHFR) gene variant (C677T) as risk factors for ischemic stroke. Recent studies have shown that hyperhomocysteinemia and MTHFR gene variant are associated with small-vessel disease, suggesting that these risk factors may underlie vascular parkinsonism, particularly in patients lacking hypertension and in those with a relatively younger age at onset of this disease.","PeriodicalId":371642,"journal":{"name":"Rinshō shinkeigaku Clinical neurology","volume":"24 1","pages":"0"},"PeriodicalIF":0.0,"publicationDate":"2016-01-21","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"116557941","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
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