Observational studyNeurology(R) neuroimmunology & neuroinflammation2026
High Relapse Rate in Anti-CASPR2 Disease: Implications for Prolonged Immunotherapy.
Observational study in Neurology(R) neuroimmunology & neuroinflammation, 2026. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 6 papers.
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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
6 citing papers in PubMed.
- Optimizing the Use of Anti-CD20 Agents in Multiple Sclerosis and Other Autoimmune Central Nervous System Disorders: A Narrative Review.Neurology and therapy · 2026Review
- Recent advances in autoantibody-mediated pain.Current opinion in supportive and palliative care · 2026Review
- Fifteen years of autoimmune encephalitis in Denmark: incidence, epidemiology, and trends in treatment.Journal of neurology · 2026Article
- The Treatment of Antibody-Mediated Encephalitis: Current, Future Therapies, Unmet Need and Patient Management.Advances in therapy · 2026Review
- A rare presentation of CASPR2-associated Morvan syndrome overlapping with GM1-positive AMSAN: a case report.Frontiers in immunology · 2026Article
- CNTNAP2: isoform- and context-specific functions in neurological disorders and cancer.Frontiers in cellular neuroscience · 2026Review
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Authors and funding
14 authors.
Funding
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Abstract
BACKGROUND AND
objectivesAnti-contactin-associated protein-like 2 (CASPR2) disease-previously considered a subtype of anti-voltage-gated potassium channel (VGKC-complex) encephalitis-is a relatively new disease entity, and information on long-term outcomes and relapses is limited. We investigated long-term clinical outcomes and factors associated with higher relapse rates. In addition, we compared different treatment strategies.
methodsIn this nationwide observational cohort study, patients with anti-CASPR2 disease were included. Clinical data were collected at diagnosis and during follow-up, both retrospectively and prospectively.
resultsForty-four patients with anti-CASPR2 disease were included (42 male patients [96%]; median age at onset 66 years [interquartile range (IQR) 61-71; range 40-86]). The median follow-up time was 55 months (IQR 38-78; range 8-200). Sixty percent of the patients experienced at least one relapse. Most patients experienced similar but fewer symptoms during relapse(s). However, new symptoms could occur during a relapse episode, mainly involving the peripheral nervous system. The median time to relapse was 10.5 months (IQR 6-14; range 3-58). Twelve of the 44 patients were diagnosed with cancer, with 3 cancers identified only at relapse. We found significantly higher anti-VGKC-complex concentrations at diagnosis ( DISCUSSION: The relapse rate in anti-CASPR2 disease is much higher than previously reported. In patients prone to relapse, (repeated) courses of rituximab appear to be most effective in preventing future relapses beyond acute therapy and tapering with oral steroids. Anti-VGKC-complex concentrations in serum can aid in monitoring of relapses and disease course in most of the patients. It is recommended to repeat tumor screening when patients relapse. CLASSIFICATION OF EVIDENCE: This study provides Class IV evidence that, in anti-CASPR2 disease, prolonged immunotherapy is associated with reduced relapse rates.
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