ArticleNeurology2019
Autoimmune gait disturbance accompanying adaptor protein-3B2-IgG.
Article in Neurology, 2019. The graph could read no effect estimate from its abstract, so it casts no vote on the map. An erratum has been issued. Cited by 27 papers.
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Who cites it
27 citing papers in PubMed.
- Placental DNA methylation captures shared and trait-specific genetic susceptibility across complex health conditions.medRxiv : the preprint server for health sciences · 2026Article
- Novel methods for neural IgG characterization and validation in autoimmune neurologic disorders.Journal of immunological methods · 2026Review
- Insights Into the Antigenic Repertoire of Unclassified Synaptic Antibodies.Annals of clinical and translational neurology · 2026Article
- Neural synaptic vesicle autoimmunity following aerosolized porcine neural tissue exposure: insights into autoimmune inflammatory polyradiculoneuropathy.EBioMedicine · 2025Article
- Acute neuronal cell death and neuroinflammation per se do not trigger secondary autoimmune encephalitis in mice.Scientific reports · 2025Article
- Case Report: Subacute combined degeneration of the spinal cord mimic accompanying adaptor protein-3B2-IgG.Frontiers in immunology · 2025Article
- Autoimmune Neurobeachin Cerebellar Ataxia.Movement disorders : official journal of the Movement Disorder Society · 2024Article
- Identification of DAGLA as an autoantibody target in cerebellar ataxia.Journal of neurology, neurosurgery, and psychiatry · 2024Article
- Autoimmune encephalitis: what the radiologist needs to know.Neuroradiology · 2024Review
- Clinical Features and Neuroimaging Findings of Neuropil Antibody-Positive Idiopathic Sporadic Ataxia of Unknown Etiology.Cerebellum (London, England) · 2023Article
- Utility of Protein Microarrays for Detection of Classified and Novel Antibodies in Autoimmune Neurologic Disease.Neurology(R) neuroimmunology & neuroinflammation · 2023Article
- The expanding spectrum of antibody-associated cerebellar ataxia: report of two new cases of anti-AP3B2 ataxia.Journal of neurology · 2023Article
- Autoimmune cerebellar ataxia associated with anti-Purkinje cells antibodies: the next frontier of neuroimmunology.Annals of translational medicine · 2023Review
- Paraneoplastic Neurologic Disorders.Current neurology and neuroscience reports · 2023Review
- Rare case of anti-CV2 paraneoplastic polyneuropathy associated with lung adenocarcinoma.BMJ case reports · 2023Article
- Therapy response in seronegative versus seropositive autoimmune encephalitis.Frontiers in immunology · 2023Article
- Rare Etiologies in Immune-Mediated Cerebellar Ataxias: Diagnostic Challenges.Brain sciences · 2022Review
- Novelties in Autoimmune and Paraneoplastic Cerebellar Ataxias: Twenty Years of Progresses.Cerebellum (London, England) · 2022Review
- Clinical features and outcome of patients with autoimmune cerebellar ataxia evaluated with the Scale for the Assessment and Rating of Ataxia.European journal of neurology · 2022Article
- Autoantibodies in neurological disease.Nature reviews. Immunology · 2021Review
Corrections and comments
- Erratum issued
Authors and funding
10 authors.
Funding
No grant is acknowledged in the PubMed record.
Abstract
objectiveTo describe phenotypes, treatment response, and outcomes of autoimmunity targeting a synaptic vesicle coat protein, the neuronal (B2) form of adaptor protein-3 (AP3).
methodsArchived serum and CSF specimens (from 616,025 screened) harboring unclassified synaptic antibodies mimicking amphiphysin-immunoglobulin G (IgG) on tissue-based indirect immunofluorescence assay (IFA) were re-evaluated for novel IgG staining patterns. Autoantigens were identified by western blot and mass spectrometry. Recombinant western blot and cell-binding assay (CBA) were used to confirm antigen specificity. Clinical data were obtained retrospectively.
resultsSerum (10) and CSF (6) specimens of 10 patients produced identical IFA staining patterns throughout mouse nervous system tissues, most prominently in cerebellum (Purkinje neuronal perikarya, granular layer synapses, and dentate regions), spinal cord gray matter, dorsal root ganglia, and sympathetic ganglia. The antigen revealed by mass spectrometry analysis and confirmed by recombinant assays (western blot and CBA) was AP3B2 in all. Of 10 seropositive patients, 6 were women; median symptom onset age was 42 years (range 24-58). Clinical information was available for 9 patients, all with subacute onset and rapidly progressive gait ataxia. Neurologic manifestations were myeloneuropathy (3), peripheral sensory neuropathy (2), cerebellar ataxia (2), and spinocerebellar ataxia (2). Five patients received immunotherapy; none improved, but they did not worsen over the follow-up period (median 36 months; range 3-94). Two patients (both with cancer) died. One of 50 control sera was positive by western blot only (but not by IFA or CBA).
conclusionAP3B2 (previously named β-neuronal adaptin-like protein) autoimmunity appears rare, is accompanied by ataxia (sensory or cerebellar), and is potentially treatable.
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