ArticleCPT: pharmacometrics & systems pharmacology2026
From Radiocopper to Cold Copper: Mechanistic Modeling and Simulation to Define Clinical Response Criteria and Biomarkers for VTX-801 in Wilson Disease.
Article in CPT: pharmacometrics & systems pharmacology, 2026. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 3 papers.
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Who cites it
3 citing papers in PubMed.
- Copper homeostasis and cuproptosis: molecular mechanisms and therapeutic opportunities.Molecular biomedicine · 2026Review
- Recent advances in research on high-frequency mutations in the ATP7B gene associated with Wilson disease in the Chinese population: from genetic evolution to precision medicine.Frontiers in molecular biosciences · 2026Review
- From Radiocopper to Cold Copper: Mechanistic Modeling and Simulation to Define Clinical Response Criteria and Biomarkers for VTX-801 in Wilson Disease.CPT: pharmacometrics & systems pharmacology · 2026Article
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4 authors.
Funding
Abstract
We developed a comprehensive, mechanistic model of human copper metabolism to support biomarker qualification for VTX-801, an adeno-associated vector-based gene therapy which is being developed to restore the mutated ATP7B copper transporter gene in Wilson disease (WD). The model integrates physiological copper kinetics with pathophysiological features of WD by distinguishing between ceruloplasmin-bound and non-ceruloplasmin-bound copper (NCC), and by explicitly incorporating ATP7B-dependent processes: biliary excretion and ceruloplasmin loading of copper. Literature-derived time-activity data from healthy subjects, heterozygous carriers, and WD patients, as well as clinical radiocopper data in plasma and feces from a pilot study in non-WD subjects, were used for model development and validation. VTX-801's dose-response was quantified in WD mouse models using ceruloplasmin oxidase activity measurement and
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