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Article: Keeping pace with the world: improving the clinical practice of anti-NMDAR encephalitis in China.

Guan, Hongzhi

2020 Volume 8, Issue 15, Page(s) 976

Language	English
Publishing date	2020-09-02
Publishing country	China
Document type	Journal Article ; Comment
ZDB-ID	2893931-1
ISSN	2305-5847 ; 2305-5839
ISSN (online)	2305-5847
ISSN	2305-5839
DOI	10.21037/atm-2020-76
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Article ; Online: Pictographs of encephalitis in Chinese characters.

Guan, Hongzhi

The Lancet. Neurology

2019 Volume 18, Issue 4, Page(s) 331

MeSH term(s)	Brain/pathology ; China ; Encephalitis/history ; History, Ancient ; Humans
Language	English
Publishing date	2019-03-11
Publishing country	England
Document type	Historical Article ; Letter
ZDB-ID	2079704-7
ISSN	1474-4465 ; 1474-4422
ISSN (online)	1474-4465
ISSN	1474-4422
DOI	10.1016/S1474-4422(19)30077-8
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Article ; Online: Research Progress in Antibodies Related to Autoimmune Encephalitis in Neuropsychiatric Systemic Lupus Erythematosus

CHEN Sixian / REN Haitao / ZENG Xiaofeng / GUAN Hongzhi

罕见病研究, Vol 1, Iss 2, Pp 217-

2022 Volume 222

Abstract: Several anti-neural antibodies are associated with neuropsychiatric systemic lupus erythematosus (NPSLE) including anti-neuronal antibodies and anti-glial cell antibodies. The anti-neuronal antibodies has two types: anti-neuronal surface protein ... ...

Abstract	Several anti-neural antibodies are associated with neuropsychiatric systemic lupus erythematosus (NPSLE) including anti-neuronal antibodies and anti-glial cell antibodies. The anti-neuronal antibodies has two types: anti-neuronal surface protein antibodies represented by anti-N-methyl-D-aspartate receptor (NMDAR) antibodies, and anti-neuronal intracellular protein antibodies. In this paper, we review and classify the anti-neural antibodies related to NPSLE.
Keywords	systemic lupus erythematosus ; central nervous system ; neuropsychiatric systemic lupus erythematosus ; anti-neural antibody ; anti-neuronal antibodies ; anti-neuronal intracellular protein antibodies ; Medicine ; R
Language	Chinese
Publishing date	2022-04-01T00:00:00Z
Publisher	Editorial Office of Journal of Rare Diseases
Document type	Article ; Online
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Article ; Online: Autoantibodies against eukaryotic translation elongation factor 1 delta in two patients with autoimmune cerebellar ataxia.

Guo, Liyuan / Ren, Haitao / Fan, Siyuan / Chao, Xingchen / Liu, Mange / Guan, Hongzhi / Wang, Jing

Frontiers in immunology

2024 Volume 14, Page(s) 1289175

Abstract: Background: Autoantibodies are useful biomarkers for the early detection and diagnosis of autoimmune cerebellar ataxia (ACA).: Objective: To identify novel autoantibody candidates in ACA patients.: Methods: Patients with cerebellar ataxia of ... ...

Abstract	Background: Autoantibodies are useful biomarkers for the early detection and diagnosis of autoimmune cerebellar ataxia (ACA). Objective: To identify novel autoantibody candidates in ACA patients. Methods: Patients with cerebellar ataxia of unknown cause were recruited from July 2018 to February 2023. Anti-neural autoantibodies in patient samples were detected by tissue-based indirect immunofluorescence assay (TBA) on rat cerebellum sections. TBA-positive samples were further screened for well-established anti-neural autoantibodies using commercial kits. Tissue-immunoprecipitation (TIP) and subsequent mass spectrometric (MS) analysis were used to explore the target antigens of autoantibodies in samples that were TBA-positive but negative for known autoantibodies. The specific binding between autoantibodies and the identified target antigen was confirmed by neutralization experiments, recombinant cell-based indirect immunofluorescence assay (CBA), and western blotting experiments. Results: The eukaryotic translation elongation factor 1 delta (EEF1D) protein was identified as a target antigen of autoantibodies in samples from a 43-year-old female ACA patient, while the specific binding of autoantibodies and EEF1D was confirmed by subsequent experiments. A second anti-EEF1D autoantibody-positive ACA patient, a 59-year-old female, was detected in simultaneous screening. The main clinical manifestations in each of the two patients were cerebellar syndrome, such as unsteady walking and limb ataxia. Both patients received immunotherapy, including corticosteroids, intravenous immunoglobulin, and mycophenolate mofetil. Their outcomes provided evidence to support the effectiveness of immunotherapy, but the cerebellar atrophy that occurred before treatment may be irreversible. Conclusion: In the current study, we identified anti-EEF1D autoantibody as a novel autoantibody candidate in ACA. Its pathological roles and diagnostic value need to be further verified in larger-scale studies.
MeSH term(s)	Female ; Humans ; Adult ; Middle Aged ; Autoantibodies ; Cerebellar Ataxia/diagnosis ; Cerebellar Ataxia/metabolism ; Cerebellar Ataxia/pathology ; Peptide Elongation Factor 1 ; Biomarkers ; Immunotherapy
Chemical Substances	Autoantibodies ; Peptide Elongation Factor 1 ; Biomarkers ; EEF1D protein, human
Language	English
Publishing date	2024-01-25
Publishing country	Switzerland
Document type	Case Reports ; Journal Article ; Research Support, Non-U.S. Gov't ; Comment
ZDB-ID	2606827-8
ISSN	1664-3224 ; 1664-3224
ISSN (online)	1664-3224
ISSN	1664-3224
DOI	10.3389/fimmu.2023.1289175
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Article ; Online: Abundant infiltration of B cells and plasma cells in brain biopsy of a male patient with severe anti-NMDA receptor encephalitis: A case report.

Wei, Linfei / Lu, Zhengjuan / Du, Zunguo / Wang, Yin / Guan, Hongzhi

Medicine

2023 Volume 102, Issue 27, Page(s) e34237

Abstract: Rationale: Anti-N-methyl-D-aspartate receptor (NMDAR) is the most common type of autoimmune encephalitis mediated by NMDAR antibodies. The pathological process remains unclear, especially in patients without tumors or infections. Autopsy and biopsy ... ...

Abstract	Rationale: Anti-N-methyl-D-aspartate receptor (NMDAR) is the most common type of autoimmune encephalitis mediated by NMDAR antibodies. The pathological process remains unclear, especially in patients without tumors or infections. Autopsy and biopsy studies have rarely been reported because of the favorable prognosis. Pathological findings generally demonstrate mild-to-moderate inflammation. This case report presents severe anti-NMDAR encephalitis in a 43-year-old man without any identified triggers. The biopsy in this patient showed extensive inflammatory infiltration with evident B cell accumulation, which enriches the pathological study of male anti-NMDAR encephalitis patients without comorbidities. Patient concerns: A 43-year-old previously healthy man presented with new-onset seizures with recurrent jerks. The initial autoimmune antibody test with serum and cerebrospinal fluid yielded negative results. After ineffective treatment for viral encephalitis, based on the imaging results indicating the possibility of diffuse glioma, the patient underwent a brain biopsy in the right frontal lobe to rule out malignancy. Diagnosis: The immunohistochemical study showed extensive inflammatory cell infiltration, consistent with the pathological changes in encephalitis. Cerebrospinal fluid and serum samples were then retested and tested positive for IgG antibodies against NMDAR. Therefore, the patient was diagnosed with anti-NMDAR encephalitis. Interventions: The patient was administered intravenous immunoglobulin (0.4 g/kg/d for 5 days), intravenous methylprednisolone (1 g/d for 5 days, 500 mg/d for 5 days, subsequently reduced to oral administration), and intravenous cyclophosphamide cycles. Outcomes: The patient developed refractory epilepsy 6 weeks later and required mechanical ventilation. Despite brief clinical improvement after extensive immunotherapy, the patient died from bradycardia and circulation. Lessons: Anti-NMDAR encephalitis cannot be ruled out even if the initial autoantibody test result is negative. For progressive encephalitis of unknown etiology, it is necessary to recheck cerebrospinal fluid for anti-NMDAR antibodies.
MeSH term(s)	Humans ; Male ; Adult ; Anti-N-Methyl-D-Aspartate Receptor Encephalitis/drug therapy ; Plasma Cells ; Immunoglobulins, Intravenous/therapeutic use ; Autoantibodies ; Biopsy ; Brain/diagnostic imaging
Chemical Substances	Immunoglobulins, Intravenous ; Autoantibodies
Language	English
Publishing date	2023-07-07
Publishing country	United States
Document type	Case Reports ; Journal Article
ZDB-ID	80184-7
ISSN	1536-5964 ; 0025-7974
ISSN (online)	1536-5964
ISSN	0025-7974
DOI	10.1097/MD.0000000000034237
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Article ; Online: Sera anti-neuronal antibodies in patients with irritable bowel syndrome and their correlations with clinical profiles.

Fan, Wenjuan / Fang, Xiucai / Fei, Guijun / Li, Xiaoqing / Guan, Hongzhi

Neurogastroenterology and motility

2023 Volume 35, Issue 11, Page(s) e14682

Abstract: Background: Immune factors were involved in the pathophysiology of irritable bowel syndrome (IBS). The aim of the study was to test anti-neuronal antibodies in sera of IBS patients and demonstrate their correlations with IBS profiles and psychological ... ...

Abstract	Background: Immune factors were involved in the pathophysiology of irritable bowel syndrome (IBS). The aim of the study was to test anti-neuronal antibodies in sera of IBS patients and demonstrate their correlations with IBS profiles and psychological disorders. Methods: Patients with IBS met Rome III criteria and excluded organic diseases were enrolled. Controls included healthy subjects (HS), slow transit functional constipation, autoimmune diseases, and so on. Indirect immunofluorescence with monkey cerebellum and small intestine as substrates was used to detect anti-neuronal antibodies including anti-cerebral neuronal antibodies (ACNA) and anti-enteric neuronal antibodies (AENA). Results: A total of 293 IBS patients, 100 HS and 153 disease controls were included in this study. The ACNA positive rate of IBS patients was significantly higher than HS (14% vs. 6%, p = 0.033). The positive rate of ACNA was significantly lower than AENA (14.0% vs. 76.8%, p = 0.028) in IBS patients. The prevalence of headache and sleeping disorder were higher in ACNA-positive IBS patients than ACNA-negative IBS patients (61% vs. 42.9%, p = 0.03; 75.6% vs. 57.1%, p = 0.03, respectively). Among IBS patients, ACNA and AENA were both negative in 21.8% patients, ACNA negative and AENA positive in 64.2% patients, and ACNA and AENA were both positive in 12.6% patients. There were no significant differences of intestinal symptoms among the three groups, while the prevalence of headache (64.9% vs. 37.5% and 44.7%, p = 0.03) and sleeping disorder (78.4% vs. 50.0% and 59.6%, p = 0.02) were higher in patients with both ACNA and AENA positive than patients with both ACNA and AENA negative, patients with ACNA negative and AENA positive. There were no significant differences of the prevalence of depression and anxiety, HAMD, and HAMA scores among the three groups. Conclusions and inferences: Anti-neuronal antibodies in sera of IBS patients were mainly targeted to enteric neurons and in a small part to cerebral neurons. ACNA were closely related to headache and sleeping disorder but unrelated to intestinal symptoms, depression, or anxiety of IBS patients.
MeSH term(s)	Humans ; Irritable Bowel Syndrome/diagnosis ; Constipation ; Autoantibodies ; Headache ; Neurons ; Surveys and Questionnaires
Chemical Substances	Autoantibodies
Language	English
Publishing date	2023-09-24
Publishing country	England
Document type	Journal Article
ZDB-ID	1186328-6
ISSN	1365-2982 ; 1350-1925
ISSN (online)	1365-2982
ISSN	1350-1925
DOI	10.1111/nmo.14682
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Zs.A 2979: Show issues

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Article ; Online: Clinical phenotypes and genetic features of hereditary transthyretin amyloidosis patients in China.

He, Xinyue / Tian, Zhuang / Guan, Hongzhi / Zhang, Shuyang

Orphanet journal of rare diseases

2022 Volume 17, Issue 1, Page(s) 337

Abstract: Background: Hereditary transthyretin amyloidosis (hATTR) is a progressive and fatal disease with heterogenous clinical presentations, limited diagnosis and poor prognosis. This retrospective analysis study aimed to report the genotypes and phenotypes of ...

Abstract	Background: Hereditary transthyretin amyloidosis (hATTR) is a progressive and fatal disease with heterogenous clinical presentations, limited diagnosis and poor prognosis. This retrospective analysis study aimed to report the genotypes and phenotypes of herediary transthyretin amyloidosis (hATTR) in Chinese through a systematic review of published literature. Methods: The systematic review included structured searches of peer-reviewed literature published from 2007 to 2020 of following online reference databases: PubMed, Web of Science and the literature database in China. Extracted data included sample size, personal information (sex, age, natural course, family history), mutation type, clinical milestones and reason of death. Results: We described 126 Chinese patients with hereditary transthyretin amyloidosis identified through a systematic review of 30 studies. The most common genotype in the Chinese population was Gly83Arg (25, 19.8%), which most likely presented visual and neurological abnormalities without reported death. The second and third most common genotypes were Val30Met (20, 15.9%) and Val30Ala (10, 7.9%). Peripheral neurological manifestations (91, 72%) were dominant in 126 patients. The followed manifestation was autonomic neurological abnormalities (73, 58%). Half of the cases were reported to have visual disorders, and nearly one-third of the cases presented cardiac abnormalities. Among all 126 reported patients, 46.03% were classified as neurological type, 30.16% as mixed type and only 2.38% as cardiac type. In addition. Chinese patients were mostly early onset, with age of onset at 41.8 (SD: 8.9) years, and the median time from onset to death was 7.5 [IQR: 5.3] years. Patients with cardiac involvement had a shorter survival duration (log Rank (Mantel-Cox), χ Conclusions: This study focused on 126 Chinese hATTR patients obtained from a literature review. A total of 26 kinds of TTR mutations were found and the most common one was Gly83Arg. As for phenotype, 46.03% were classified as neurological type, 30.16% as mixed type and only 2.38% as cardiac type. Chinese hATTR patients were mostly early onset (AO 41.8 years), and the median time from onset to death was 7.5 years.
MeSH term(s)	Amyloid Neuropathies, Familial/genetics ; Humans ; Phenotype ; Prealbumin/genetics ; Retrospective Studies
Chemical Substances	Prealbumin
Language	English
Publishing date	2022-09-02
Publishing country	England
Document type	Journal Article ; Systematic Review ; Research Support, Non-U.S. Gov't
ZDB-ID	2225857-7
ISSN	1750-1172 ; 1750-1172
ISSN (online)	1750-1172
ISSN	1750-1172
DOI	10.1186/s13023-022-02481-9
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Article ; Online: Pitfalls in the diagnosis of herpes simplex virus-1 encephalitis: case report.

Wei, Yanping / Guan, Hongzhi

Neurological sciences : official journal of the Italian Neurological Society and of the Italian Society of Clinical Neurophysiology

2020 Volume 42, Issue 3, Page(s) 1189–1191

MeSH term(s)	Antiviral Agents/therapeutic use ; Encephalitis, Herpes Simplex/diagnosis ; Encephalitis, Herpes Simplex/drug therapy ; Humans ; Simplexvirus
Chemical Substances	Antiviral Agents
Language	English
Publishing date	2020-09-29
Publishing country	Italy
Document type	Case Reports ; Letter
ZDB-ID	2016546-8
ISSN	1590-3478 ; 1590-1874
ISSN (online)	1590-3478
ISSN	1590-1874
DOI	10.1007/s10072-020-04765-z
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Zs.A 1877: Show issues

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Article ; Online: Seizure semiology and predictors of outcomes in Chinese patients with glutamic acid decarboxylase antibody-associated neurological syndrome.

Lin, Nan / Bai, Lin / Liu, Qing / Chen, Jianhua / Ren, Haitao / Guan, Hongzhi / Lu, Qiang

BMC neurology

2023 Volume 23, Issue 1, Page(s) 149

Abstract: Background: In the current study, seizure semiology and potential predictive factors of seizure outcomes in glutamic acid decarboxylase antibody (GAD Ab)-associated neurological syndrome were investigated.: Methods: In this study, 32 Chinese patients ...

Abstract	Background: In the current study, seizure semiology and potential predictive factors of seizure outcomes in glutamic acid decarboxylase antibody (GAD Ab)-associated neurological syndrome were investigated. Methods: In this study, 32 Chinese patients with GAD Ab-associated neurological syndrome who presented with seizures at Peking Union Medical College Hospital from January 2017 to October 2022 were reviewed; 30 had a follow-up duration of more than 1 year. Results: Among the 32 patients, 10 presented with epilepsy alone. Concomitant neurological syndromes were observed in 22 patients, including limbic encephalitis (n = 20), stiff-person syndrome (SPS, n = 1), and cerebellar ataxia (n = 1). Bilateral tonic-clonic seizures were observed in 21 patients (65.6%). Focal seizures occurred in 27 patients (84.4%); 17 had focal motor seizures and 18 focal non-motor seizures. Among 30 patients with long-term follow-up, 11 (36.7%) were seizure-free. Acute/subacute onset (p = 0.049) and comorbidity of limbic encephalitis with epilepsy (p = 0.023) led to better seizure outcomes. Patients with persistent epilepsy were more likely to have focal seizure (p = 0.003) and higher frequency of seizure (p = 0.001). Furthermore, these patients tended to have longer intervals from onset to immunomodulatory treatments. Early immunotherapy (within 6 months from onset) was administered in 81.8% of seizure-free patients but only in 42.1% of patients with persistent seizures. However, steroid and immunosuppressant duration did not differ in the two groups. Repeated serum GAD Ab tests during the follow-up showed no association with seizure outcomes. Conclusions: The seizure manifestations are diverse and variable. Approximately one third of patients achieved seizure remission during long-term follow-up. The type and frequency of seizures may influence the seizure outcomes. Early immunotherapy, especially within 6 months, may lead to better seizure outcomes.
MeSH term(s)	Humans ; Glutamate Decarboxylase ; Anticonvulsants/therapeutic use ; Limbic Encephalitis ; East Asian People ; Seizures/drug therapy ; Epilepsy/drug therapy ; Antibodies
Chemical Substances	Glutamate Decarboxylase (EC 4.1.1.15) ; Anticonvulsants ; Antibodies
Language	English
Publishing date	2023-04-11
Publishing country	England
Document type	Journal Article
ZDB-ID	2041347-6
ISSN	1471-2377 ; 1471-2377
ISSN (online)	1471-2377
ISSN	1471-2377
DOI	10.1186/s12883-023-03182-x
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Article ; Online: Isolated Myoclonus of the Vocal Folds in Alexander Disease.

Fan, Siyuan / Zhao, Yang / Guan, Hongzhi

JAMA neurology

2021 Volume 79, Issue 2, Page(s) 197

MeSH term(s)	Alexander Disease/complications ; Alexander Disease/physiopathology ; Female ; Gait Disorders, Neurologic ; Glial Fibrillary Acidic Protein/genetics ; Humans ; Hypotension, Orthostatic ; Laryngoscopy ; Middle Aged ; Myoclonus/etiology ; Myoclonus/physiopathology ; Vocal Cords/physiopathology
Chemical Substances	GFAP protein, human ; Glial Fibrillary Acidic Protein
Language	English
Publishing date	2021-12-20
Publishing country	United States
Document type	Case Reports ; Journal Article
ZDB-ID	2702023-X
ISSN	2168-6157 ; 2168-6149
ISSN (online)	2168-6157
ISSN	2168-6149
DOI	10.1001/jamaneurol.2021.4707
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