ArticleNeurology. Clinical practice2021
CASPR2-Related Morvan Syndrome: Autonomic, Polysomnographic, and Neuropsychological Observations.
Article in Neurology. Clinical practice, 2021. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 13 papers, 1 of them a synthesis that pooled it.
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Who cites it
13 citing papers in PubMed, 1 synthesis or guideline pooled it, 27 citations in OpenAlex.
- Contactin-associated protein-like 2 antibody-associated autoimmune encephalitis in children: case reports and systematic review of literature.Acta neurologica Belgica · 2023Pooled it
- Morvan syndrome in a patient with progressing metastatic thymoma and myasthenia gravis.BMJ case reports · 2026Article
- CASPR2 antibody-related neurological syndromes in children: three cases report and literature review.BMC pediatrics · 2026Review
- A Rare Adolescent Presentation of Morvan Syndrome: Diagnostic Challenges and Therapeutic Response.Clinical case reports · 2026Article
- Expanding the clinical spectrum of pediatric CASPR2 antibody-associated autoimmune encephalitis: a multicenter case series.Frontiers in immunology · 2026Article
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- Article
- Thymectomy for Morvan Syndrome Associated With Thymoma.Annals of thoracic surgery short reports · 2025Article
- Neuromyotonia and CASPR2 Antibodies: Electrophysiological Clues to Disease Pathophysiology.Biomolecules · 2025Review
- Cardiovascular Autonomic Dysfunction Before and After Chemotherapy in Cancer Patients.Journal of clinical neurology (Seoul, Korea) · 2024Review
- Anti-contactin-associated protein-like 2 antibody autoimmune encephalitis with rapidly progressive parkinsonism: a case report and literature review.Acta neurologica Belgica · 2023Review
- Article
- Painful Itch in a Fidgety Girl.Annals of Indian Academy of NeurologyArticle
Corrections and comments
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Authors and funding
15 authors at 1 institution in 1 country.
Funding
No grant is acknowledged in the PubMed record.
Abstract
objectiveMorvan syndrome is characterized by central, autonomic, and peripheral hyperexcitability due to contactin-associated protein 2 (CASPR2) antibody. Our objective was to study the clinical spectrum, electrophysiologic, autonomic, polysomnographic, and neuropsychological profile in patients with CASPR2-related Morvan syndrome.
methodsSerum and CSF samples that were CASPR2 antibody positive from 2016 to 2019 were assessed. Among them, patients with Morvan syndrome diagnosed based on clinical and electrophysiologic basis were included.
resultsFourteen (M:F = 10:4) patients with Morvan syndrome were included with age at onset of 37.1 ± 17.5 years. The clinical features were muscle twitching (12), insomnia (12), pain (11), paresthesias (9), hyperhidrosis (7), hypersalivation (6), double incontinence (3), spastic speech (2), dysphagia (2), behavioral disturbances (2), seizures (1), and cold intolerance (1). Neurologic examination revealed myokymia (12), hyperactive tendon reflexes (10), and tremor (6). EMG revealed neuromyotonia (12) and increased spontaneous activity (7). Autonomic function tests conducted in 8 patients revealed definite autonomic dysfunction (4), orthostatic hypotension (2), early dysfunction (1), and postural orthostatic tachycardia syndrome (1). Polysomnography findings in 6 patients revealed insomnia (3), absence of deep sleep (1), high-frequency beta activity (1), REM behavior disorder (1), and periodic leg movements (1). Neuropsychological evaluation showed subtle involvement of the left frontal and temporal lobe. Malignancy workup was negative. All patients were treated with steroids. There was complete neurologic resolution in follow-up with persistent neuropathic pain in 5 patients.
conclusionsThis study has contributed to the growing knowledge on CASPR2-related Morvan syndrome. It is important for an increased awareness and early recognition as it is potentially treatable by immunotherapy.
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.