ArticleJournal of neurology2023
Long-term outcome of patients with neurological form of Wilson's disease compliant to the de-coppering treatment.
Article in Journal of neurology, 2023. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 9 papers.
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Who cites it
9 citing papers in PubMed, 12 citations in OpenAlex.
- Emotion-tremor coupling in Wilson's disease: EEG microstate C as a marker of salience network dysregulation.Orphanet journal of rare diseases · 2026Article
- Disorders Mimicking Wilson's Disease: Clinical, Biochemical, and Molecular Perspectives for Accurate Differential Diagnosis.Diagnostics (Basel, Switzerland) · 2026Review
- Wilson's disease: does a neuropsychiatric phenotype without liver involvement truly exist?Neurological sciences : official journal of the Italian Neurological Society and of the Italian Society of Clinical Neurophysiology · 2026Article
- Improvement in sleep following treatment initiation in newly diagnosed, treatment-naïve patients with Wilson's disease.Neurological sciences : official journal of the Italian Neurological Society and of the Italian Society of Clinical Neurophysiology · 2025Article
- Clinical Determinants and Prognostic Impact of Delayed Diagnosis in Wilson's Disease.Diagnostics (Basel, Switzerland) · 2025Article
- Correlation between neuroimaging, neurological phenotype, and functional outcomes in Wilson's disease.Neurological sciences : official journal of the Italian Neurological Society and of the Italian Society of Clinical Neurophysiology · 2024Article
- The physiological and pathophysiological roles of copper in the nervous system.The European journal of neuroscience · 2024Review
- Assessment of sleep disturbance in patients with Wilson's disease.BMC psychiatry · 2024Article
- Clinical Spectrum, Radiological Correlation and Outcome of Movement Disorders in Wilson's Disease.Tremor and other hyperkinetic movements (New York, N.Y.) · 2023Article
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Authors and funding
12 authors at 2 institutions in 1 country.
Funding
No grant is acknowledged in the PubMed record.
Abstract
backgroundA substantial proportion of Wilson's disease (WD) patients exhibit residual neurological symptoms. Data on the prognostic value of initial clinical features and treatment choices in WD patients compliant to the therapy is relatively sparse.
aimThe aim of the present study was to identify predictors of the long-term outcome of patients with WD with good treatment adherence.
methodsForty patients with neurological form of WD were evaluated before the de-coppering treatment initiation (based on the medical records) and after mean 15.25 ± 11.24 years of the stable treatment. Severity of neurological symptoms were assessed with a tier two of Global Assessment Scale (GAS) for Wilson's Disease.
resultsThe most frequent symptoms prior to treatment initiation were dysarthria (90%), tremor (90%), clumsiness (67.5%), depression (67.5%), and gait disturbance (62.5%). Significant decrease in the frequency of dysarthria, clumsiness, tremor, gait disturbance, postural instability and an improvement in school/work performance were observed after the long-term treatment, while frequency of dysphagia, drooling, bradykinesia and rigidity, dystonic and choreatic features did not change. Overall symptom severity decreased over time. Presence of dystonia before treatment initiation was the only identified predictor of worse residual GAS score. Greater severity of residual dystonia was associated with female gender and longer disease duration.
conclusionAlthough patients with neurological form of WD compliant to de-coppering treatment had favorable disease outcome, a significant burden of residual neurological symptoms was observed after the long-term follow-up. Dystonia at disease onset was the only identified predictor of the worse long-term outcome.
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