GFAP IgG associated inflammatory polyneuropathy

Paul, Pritikanta; McKeon, Andrew; Pittock, Sean J.; Klein, Christopher J.; Shah, S. M.; Toledano, Michel; Mills, John R.; Dubey, Divyanshu

doi:10.1016/j.jneuroim.2020.577233

Cited by 17 publications

(24 citation statements)

References 15 publications

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“…Finally, we found frequent PNS involvement, as described recently. 17 This included axonal large fiber polyneuropathy for about half of the cases and cranial or radicular neuropathies associated with meningoencephalomyelitis. We also identified a genuine paraneoplastic sensitive neuronopathy with meningitis and a symptomatic case of multiple involvement of the cranial nerves.…”

Section: Discussionmentioning

confidence: 99%

Glial Fibrillary Acidic Protein Autoimmunity

Dumonceau

Améli²,

Rogemond³

et al. 2022

Neurology

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Objectives:To report the clinical, biological, imaging features, and the clinical course of a French cohort of patients with glial fibrillar acidic protein (GFAP) autoantibodies.Methods:We retrospectively included all patients tested positive for GFAP antibodies in the cerebrospinal fluid, by immunohistochemistry and confirmed by cell-based assay using cells expressing human GFAPα, since 2017, from two French referral centers.Results:We identified 46 patients with GFAP antibodies. Median age at onset was 43 years, and 65% were men. Infectious prodromal symptoms were found in 82%. Other auto-immune diseases were found in 22% of patients, and coexisting neural autoantibodies in 11%. Tumors were present in 24%, and T cell dysfunction in 23%. The most frequent presentation was subacute meningoencephalitis (85%) with cerebellar dysfunction in 57% of cases. Other clinical presentation included myelitis (30%), visual (35%) and peripheral nervous system involvement (24%). MRI showed perivascular radial enhancement in 32%, periventricular T2 hyperintensity in 41%, brainstem involvement in 31%, leptomeningeal enhancement in 26%, and reversible splenial lesions in 4 cases. 33/40 patients had a monophasic course, associated to a good outcome at last follow-up (Rankin Score≤2: 89%), despite a severe clinical presentation. Adult and pediatric features are similar. Thirty-two patients were treated with immunotherapy. 11/22 patients showed negative conversion of GFAP antibodies.Interpretation:GFAP auto-immunity is mainly associated with acute/subacute meningoencephalomyelitis with prodromal symptoms, for which tumors and T cell dysfunction are frequent triggers. The majority of patients followed a monophasic course with a good outcome.

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Section: Discussionmentioning

confidence: 99%

Glial Fibrillary Acidic Protein Autoimmunity

Dumonceau

Améli²,

Rogemond³

et al. 2022

Neurology

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“…[40] It is generally linked with CNS autoimmune diseases with occasional reports of peripheral neuropathy. [62] Independent research groups have reported NO as an essential nociceptive facilitator that has an central role in chronic pain conditions at central and also peripheral levels. [63,64] It is synthesized by three isoforms of nitric oxide synthase (NOS) and is associated with the progress and preservation of the neuropathic pain.…”

Section: Discussionmentioning

confidence: 99%

Protective effect of naringin against oxaliplatin‐induced peripheral neuropathy in rats: A behavioral and molecular study

Semis

Kandemir

Çağlayan

et al. 2022

J Biochem & Molecular Tox

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Oxaliplatin (OXL) is a chemotherapeutic drug used for metastatic and other types of cancer, but it causes peripheral neuropathy as a dose‐limiting side effect. Herein, we used the rat model of OXL‐induced peripheral neuropathy to demonstrate the protective effects of naringin (NRG) in this neuropathy. In this study, rats were injected with OXL (4 mg/kg, body weight, i.p.) in 5% glucose solution 30 min after oral administration of NRG (50 and 100 mg/kg, body weight) on the 1st, 2nd, 5th, and 6th days. OXL caused sensory and motor neuropathy (as revealed by the hot plate, tail flick, rota‐rod, and cold hyperalgesia tests) in the sciatic nerve of rats. Coadministration of oral NRG alleviated OXL‐induced sensory and motor neuropathy. Levels of superoxide dismutase, catalase, glutathione peroxidase, nuclear factor erythroid 2‐related factor 2, Heme oxygenase‐1, nuclear factor‐κ B, tumor necrosis factor‐α, interleukin‐1β, Bax, Bcl‐2, caspase‐3, paraoxonase, mitogen‐activated protein kinase 14, neuronal nitric oxide synthase (nNOS), acetylcholinesterase, and arginase 2 in the sciatic nerve tissues were assessed by real‐time polymerase chain reaction. Moreover, the protein levels of caspase‐3, Bax, Bcl‐2, intercellular adhesion molecules‐1, glial fibrillary acidic protein, and nNOS were examined by Western blot analysis. NRG treatment significantly improved all the above‐mentioned parameters and reduced OXL‐induced oxidative stress, inflammation, and apoptosis in the sciatic nerve tissue. In conclusion, this study demonstrated that NRG significantly attenuated OXL‐induced peripheral neuropathy and might be considered as a new protective agent to prevent the OXL‐induced peripheral neuropathy.

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“…More recently, several studies have reported GFAP-IgG-positive patients who presented with peripheral neuropathy (PN). 3 – 5 However, the frequency and phenotypes of PN associated with GFAP-IgG have not been well described.…”

Section: Introductionmentioning

confidence: 99%

Clinical and electrophysiological characteristics of peripheral neuropathy in autoimmune glial fibrillary acidic protein astrocytopathy: an observational study and literature review

Deng

Wang

Qiu

et al. 2023

Ther Adv Neurol Disord

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Background: The phenotype of peripheral neuropathy (PN) associated with glial fibrillary acidic protein-immunoglobulin G (GFAP-IgG) has not been well described. Objectives: The aim of this study was to report the frequency, clinical, and electrophysiological characteristics of PN in GFAP-IgG-positive patients. Design: This study is a single-center retrospective observational study. Data Sources and methods: GFAP-IgG-positive patients with PN were retrospectively identified from the Huashan Hospital Autoimmune Encephalitis Cohort between 2017 and 2021. Eight patients who presented with PN from other published studies were also included in the analysis. The clinical and electrophysiological characteristics of GFAP-IgG-related PN were described. Results: A total of 21 (31%) patients (7 females, 14 males; Mage: 42 ± 16 years) from a cohort of 68 GFAP-IgG-positive patients presented with PN. Twenty of 21 patients had symmetrical weakness. Sensory and autonomic symptoms were present in 16 and 15 patients, respectively. Lower extremities were the most frequently involved regions for both motor (20/21) and sensory (15/21) symptoms. Moreover, 13 patients (4 females, 9 males; Mage: 43 ± 13 years) had electrodiagnostic study data, and 12 of 13 patients had abnormal findings. Regarding clinical features, motor nerve fibers were predominantly involved (12/13), and symmetric lower extremities (12/13) were the most commonly affected regions. Axonal neuropathy is the typical underlying pathophysiologic process of PN. All 21 patients responded to immunotherapy. However, four patients with tetraplegia had poor outcomes, and PN was the major determinant of their long-term disability. Most cases (6/8) from the literature presented with similar clinical and electrophysiological features to those from our cohort. Conclusion: Peripheral nerves could be involved in autoimmune GFAP astrocytopathy. Predominant motor axonal neuropathy mainly involving the lower extremities is the most common PN phenotype in this disorder. GFAP-IgG-related PN is responsive to immunotherapy. Registration: Chinese Clinical Trial Registry: ChiCTR2000029115 ( http://www.chictr.org ).

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GFAP IgG associated inflammatory polyneuropathy

Cited by 17 publications

References 15 publications

Glial Fibrillary Acidic Protein Autoimmunity

Glial Fibrillary Acidic Protein Autoimmunity

Protective effect of naringin against oxaliplatin‐induced peripheral neuropathy in rats: A behavioral and molecular study

Clinical and electrophysiological characteristics of peripheral neuropathy in autoimmune glial fibrillary acidic protein astrocytopathy: an observational study and literature review

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