ArticleAnnals of clinical and translational neurology2024
Adaptive behavior assessed by Vineland-3 as comprehensive outcome measure in vanishing white matter.
Article in Annals of clinical and translational neurology, 2024. 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.
- Efficacy and safety of allogeneic umbilical cord blood mononuclear cells therapy in children with autism spectrum disorder and immune dysregulation: a single-center, double-blinded, randomized, placebo-controlled trial.Stem cell research & therapy · 2026Trial
- Optimizing developmental outcomes in premature infants through a multidisciplinary approach to care.Pediatric research · 2026Article
- Consensus-Based Expert Recommendations for Diagnosis and Clinical Management of Vanishing White Matter.Neurology · 2025Review
- Functional ability profiles in beta-propeller protein-associated neurodegeneration (BPAN).Molecular genetics and metabolism · 2025Observational
- ARSA Variants Associated With Cognitive Decline and Long-Term Preservation of Motor Function in Metachromatic Leukodystrophy.Journal of inherited metabolic disease · 2025Observational
- Cognitive and Adaptive Functioning of CTNNB1 Syndrome Patients: A Comparison With Autism Spectrum Disorder and Cerebral Palsy.Journal of intellectual disability research : JIDR · 2025Article
Corrections and comments
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Authors and funding
8 authors.
Funding
Abstract
objectivesInvestigate the results and usability of the Vineland-3 as an outcome measure in vanishing white matter patients.
methodsA cross-sectional investigation of the Vineland-3 based on interviews with caregivers, the Health Utilities Index, and the modified Rankin Scale in 64 vanishing white matter patients.
resultsAdaptive behavior measured with the Vineland-3 is impaired in the vast majority of vanishing white matter patients and significantly impacts daily life. Typically, the daily living skills domain is most severely affected and the socialization domain is the least affected. Based on the metric properties and the clinical relevance, the standard scores for the daily living skills domain and Adaptive Behavior Composite have the best properties to be used as an outcome measure.
interpretationThe Vineland-3 appears to be a useful outcome measure to explore and quantify complex cognitive, behavioral, and psychiatric impairments affecting daily functioning in vanishing white matter. Further research should address the longitudinal evaluation of this tool and its additional value to standard neuropsychological and clinical examination.
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Registered trials
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