ArticleNeurology(R) neuroimmunology & neuroinflammation2018
Glycine receptor modulating antibody predicting treatable stiff-person spectrum disorders.
Article in Neurology(R) neuroimmunology & neuroinflammation, 2018. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 39 papers, 2 of them syntheses that pooled it.
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The trial behind it
Trials whose registry record cites this paper, or whose number appears in the abstract. A trial that started after this paper was published is citing it as background, not reporting it.
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Who cites it
39 citing papers in PubMed, 2 syntheses or guidelines pooled it, 83 citations in OpenAlex.
- Progressive Encephalomyelitis With Rigidity and Myoclonus With Glycine Receptor Antibodies: Clinical Features and Outcomes.Neurology(R) neuroimmunology & neuroinflammation · 2025Pooled it
- Cerebrospinal Fluid Findings in Patients With Autoimmune Encephalitis-A Systematic Analysis.Frontiers in neurology · 2019Pooled it
- Long-Term Outcomes in Stiff Person Spectrum Disorder.European journal of neurology · 2026Article
- Testing for neural antibodies in autoimmune encephalitis: who, what, where, when, why, and how.Therapeutic advances in neurological disorders · 2026Review
- Case report: Atypical anti-GlyR antibody-positive pediatric progressive encephalomyelitis with rigidity and myoclonus presenting as chronic joint pain.Frontiers in immunology · 2026Article
- Polyclonal glycine receptor aAbs: a challenge for personalized epitope characterization.Frontiers in molecular neuroscience · 2026Article
- Autologous hematopoietic stem cell transplantation in a patient with multi-refractory stiff person syndrome.Bone marrow transplantation · 2025Article
- Ten Years ofNeurology(R) neuroimmunology & neuroinflammation · 2025Article
- Population-Based Study of the Epidemiology of Stiff Person Syndrome in a Large Colorado-Based Health System.Neurology · 2024Article
- Comprehensive Analysis of Paraneoplastic Neurologic Syndrome and PNS-CARE Diagnostic Criteria in Clinical Practice.Neurology(R) neuroimmunology & neuroinflammation · 2024Article
- Stiff-person syndrome and related disorders - diagnosis, mechanisms and therapies.Nature reviews. Neurology · 2024Review
- Autoimmune central nervous system disorders: Antibody testing and its clinical utility.Clinical biochemistry · 2024Review
- Successful Autologous Hematopoietic Stem Cell Transplant in Glycine Receptor Antibody-Positive Stiff Person Syndrome: A Case ReportNeurology(R) neuroimmunology & neuroinflammation · 2024Article
- Management of Paraneoplastic Syndromes in the Era of Immune Checkpoint Inhibitors.Current treatment options in oncology · 2024Review
- Paraneoplastic progressive encephalomyelitis with rigidity and myoclonus associated with monoclonal B-cell lymphocytosis in the setting of longstanding methotrexate use: case report.Frontiers in immunology · 2024Article
- Clinical features in antiglycine receptor antibody-related disease: a case report and update literature review.Frontiers in immunology · 2024Review
- Prevalence, Clinical Profiles, and Prognosis of Stiff-Person Syndrome in a Japanese Nationwide Survey.Neurology(R) neuroimmunology & neuroinflammation · 2023Article
- Immunotherapy-Responsive Neuropathic Pain and Allodynia in a Patient With Glycine Receptor Autoantibodies: A Case Report.Neurology(R) neuroimmunology & neuroinflammation · 2023Article
- Eculizumab for the treatment of glycine receptor antibody associated stiff-person syndrome.Journal of neurology · 2023Article
- Stiff person spectrum disorder diagnosis, misdiagnosis, and suggested diagnostic criteria.Annals of clinical and translational neurology · 2023Article
Corrections and comments
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Authors and funding
9 authors at 1 institution in 1 country.
Funding
No grant is acknowledged in the PubMed record.
Abstract
backgroundGlycine receptor alpha-1 subunit (GlyRα1)-immunoglobulin G (IgG) is diagnostic of stiff-person syndrome (SPS) spectrum but has been reported detectable in other neurologic diseases for which significance is less certain.
methodsTo assess GlyRα1-IgGs as biomarkers of SPS spectrum among patients and controls, specimens were tested using cell-based assays (binding [4°C] and modulating [antigen endocytosing, 37°C]). Medical records of seropositive patients were reviewed.
resultsGlyRα1-IgG (binding antibody) was detected in 21 of 247 patients with suspected SPS spectrum (8.5%) and in 8 of 190 healthy subject sera (4%) but not CSF. Among 21 seropositive patients, 20 had confirmed SPS spectrum clinically, but 1 was later determined to have a functional neurologic disorder. Sera from 9 patients with SPS spectrum , but not 7 controls, nor the functional patient, caused GlyRα1 modulation (100% specificity). SPS spectrum phenotypes included progressive encephalomyelitis with rigidity and myoclonus (PERM) (8), classic SPS (5), stiff limb (5), stiff trunk (1), and isolated exaggerated startle (hyperekplexia, 1). Neuropsychiatric symptoms present in 12 patients (60%) were anxiety (11), depression (6), and delirium (3). Anxiety was particularly severe in 3 patients with PERM. Objective improvements in SPS neurologic symptoms were recorded in 16 of 18 patients who received first-line immunotherapy (89%, 9/10 treated with corticosteroids, 8/10 treated with IVIg, 3/4 treated with plasma exchange, and 1 treated with rituximab). Treatment-sparing maintenance strategies were successful in 4 of 7 patients (rituximab [2/3], azathioprine [1/1], and mycophenolate [1/3]).
conclusionsGlyRα1-modulating antibody improves diagnostic specificity for immunologically treatable SPS spectrum disorders. CLASSIFICATION OF EVIDENCE: This study provides Class IV evidence that GlyRα1-modulating antibody accurately identifies patients with treatable SPS spectrum disorders.
Identifiers
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Registered trials
Read under generation 80e0d062 · epoch 390. Bibliography from PubMed, PubMed Central and OpenAlex; grants from NIH RePORTER; trial links from ClinicalTrials.gov; estimates, votes and beliefs from the OpenQuestion graph.