Glial fibrillary acidic protein (GFAP) associated autoimmune meningoencephalitis in a patient receiving nivolumab

Kapadia, Ronak K.; Ney, Douglas; Hannan, Markus; Farley, Morgan; Pastula, Daniel M.; Piquet, Amanda L.

doi:10.1016/j.jneuroim.2020.577259

Cited by 13 publications

(7 citation statements)

References 13 publications

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“…Regarding autoimmune antibodies of AIE related to ICIs, antibodies against intracellular antigens (anti-Ma2, anti-Hu, anti-GAD, an unspecified Purkinje cell antibody, anti-Ri, anti-GFAP) were more frequent than those against cell-surface antigens (anti-NMDA receptor and anti-CASPR2) (4,6,8,(16)(17)(18)(19)(20)(21)(22). As an autoimmune antibody against intracellular antigens, the anti-GAD65 antibody is associated with several neurological syndromes, including stiff-person syndrome, cerebellar ataxia, and epilepsy (3,(23)(24)(25).…”

Section: Discussionmentioning

confidence: 99%

Anti-GAD65 Antibody-Associated Autoimmune Encephalitis With Predominant Cerebellar Involvement Following Toripalimab Treatment: A Case Report of a Novel irAE of Toripalimab

et al. 2022

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Toripalimab (Junshi Bioscience Co., Ltd) is a new immune checkpoint inhibitor (ICI) that targets programmed cell death protein 1 (PD-1) in various cancers, including metastatic melanoma. No neurological immune-related adverse events (n-irAEs) of toripalimab have been reported, except for neuromuscular involvement. We report a case of a 63-year-old woman who presented with severe vertigo, vomiting, nystagmus, cerebellar ataxia, and cognitive impairment after toripalimab treatment for metastatic melanoma. Compared with the concomitant cognitive dysfunction and a pathological reflex involving the cerebral cortex, the signs and symptoms of cerebellar involvement were much more prominent. Anti-glutamic acid decarboxylase 65 (anti-GAD65) antibody was positive in both serum and cerebrospinal fluid (CSF). After intravenous immunoglobulin (IVIG) and methylprednisolone (IVMP) administration, the symptoms of vertigo and vomiting resolved, with cognitive impairment and cerebellar ataxia remaining. This is the first report of autoimmune encephalitis (AIE) as an n-irAE of toripalimab.

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Section: Discussionmentioning

confidence: 99%

Anti-GAD65 Antibody-Associated Autoimmune Encephalitis With Predominant Cerebellar Involvement Following Toripalimab Treatment: A Case Report of a Novel irAE of Toripalimab

et al. 2022

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“…Autoimmune GFAP meningoencephalitis has rarely been reported in a patient after viral infection 7 or treatment with immune checkpoint inhibitors. 8 Most patients with GFAP antibodies in the CSF were responsive to immunotherapy, regardless of typical or atypical clinical features. [5][6][7][8] Although the present case had novel clinical and radiological phenotypes, responsiveness to immunotherapy was consistent with previous findings.…”

Section: Discussionmentioning

confidence: 97%

“…8 Most patients with GFAP antibodies in the CSF were responsive to immunotherapy, regardless of typical or atypical clinical features. [5][6][7][8] Although the present case had novel clinical and radiological phenotypes, responsiveness to immunotherapy was consistent with previous findings.…”

Section: Discussionmentioning

confidence: 97%

Parainfectious Anti-Glial Fibrillary Acidic Protein-Associated Meningoencephalitis

Joo

Yoo²,

Ahn

2022

JMD

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Movement disorders associated with glial fibrillary acidic protein (GFAP) autoantibodies have rarely been reported as ataxia or tremors. A 32-year-old man with headache and fever, initially diagnosed with viral meningoencephalitis, showed gradual improvement with empirical treatment. Two weeks after the illness, he suddenly developed orofacial, tongue, and neck dyskinesia accompanied by oculomotor abnormalities, which developed into severe generalized choreoballism. Brain magnetic resonance imaging (fluid-attenuated inversion recovery) showed signal hyperintensities in the bilateral globus pallidus interna. The clinical picture suggested an acute inflammatory trigger of secondary autoimmune encephalitis. The autoimmune antibody test was positive for GFAP, with the strongest reactivity in the cerebrospinal fluid (CSF) before treatment and decreased reactivity in serial CSF examinations during immunotherapy. Dyskinesia gradually improved to the extent that it could be controlled with only oral medications. This patient presented with parainfectious GFAP meningoencephalitis with distinctive clinical features and imaging findings.

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“…Hypoglycorrhachia can also be observed in neurosarcoidosis, but the absence of sarcoidosis signs and symptoms on physical examination, laboratory studies, body imaging acutely, and longitudinal yearly followup were inconsistent with this diagnosis. Low CSF glucose has also been reported in autoimmune meningoencephalitis due to checkpoint inhibitor therapy ( 15 – 17 ) and GFAP meningoencephalitis ( 18 )—the most common known autoantigen associated with autoimmune meningoencephalits ( 11 ). Although we did not test for GFAP antibodies by CBA, our case did not show GFAP-like staining by TBIF.…”

Section: Discussionmentioning

confidence: 99%

Dual ankyrinG and subpial autoantibodies in a man with well-controlled HIV infection with steroid-responsive meningoencephalitis: A case report

et al. 2023

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Neuroinvasive infection is the most common cause of meningoencephalitis in people living with human immunodeficiency virus (HIV), but autoimmune etiologies have been reported. We present the case of a 51-year-old man living with HIV infection with steroid-responsive meningoencephalitis whose comprehensive pathogen testing was non-diagnostic. Subsequent tissue-based immunofluorescence with acute-phase cerebrospinal fluid revealed anti-neural antibodies localizing to the axon initial segment (AIS), the node of Ranvier (NoR), and the subpial space. Phage display immunoprecipitation sequencing identified ankyrinG (AnkG) as the leading candidate autoantigen. A synthetic blocking peptide encoding the PhIP-Seq-identified AnkG epitope neutralized CSF IgG binding to the AIS and NoR, thereby confirming a monoepitopic AnkG antibody response. However, subpial immunostaining persisted, indicating the presence of additional autoantibodies. Review of archival tissue-based staining identified candidate AnkG autoantibodies in a 60-year-old woman with metastatic ovarian cancer and seizures that were subsequently validated by cell-based assay. AnkG antibodies were not detected by tissue-based assay and/or PhIP-Seq in control CSF (N = 39), HIV CSF (N = 79), or other suspected and confirmed neuroinflammatory CSF cases (N = 1,236). Therefore, AnkG autoantibodies in CSF are rare but extend the catalog of AIS and NoR autoantibodies associated with neurological autoimmunity.

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Glial fibrillary acidic protein (GFAP) associated autoimmune meningoencephalitis in a patient receiving nivolumab

Cited by 13 publications

References 13 publications

Anti-GAD65 Antibody-Associated Autoimmune Encephalitis With Predominant Cerebellar Involvement Following Toripalimab Treatment: A Case Report of a Novel irAE of Toripalimab

Anti-GAD65 Antibody-Associated Autoimmune Encephalitis With Predominant Cerebellar Involvement Following Toripalimab Treatment: A Case Report of a Novel irAE of Toripalimab

Parainfectious Anti-Glial Fibrillary Acidic Protein-Associated Meningoencephalitis

Dual ankyrinG and subpial autoantibodies in a man with well-controlled HIV infection with steroid-responsive meningoencephalitis: A case report

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