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Prospective Pilot Study of a Single Daily Dosage of Trientine for the Treatment of Wilson Disease
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Prospective Pilot Study of a Single Daily Dosage of Trientine for the Treatment of Wilson Disease
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Journal Article
Prospective Pilot Study of a Single Daily Dosage of Trientine for the Treatment of Wilson Disease
2015
Overview
Background
Wilson disease requires lifelong therapy, currently given daily in multiple divided dosages.
Aim
To prospectively evaluate once-daily trientine as therapy for Wilson disease.
Methods
Study group: eight patients (seven males) aged 22–71 years with stable Wilson disease treated from 4 to 50 years. Patients were monitored for 3 months then for 12 months on a single daily dose of trientine (15 mg/kg).
Results
All patients remained clinically well. ALT and AST fluctuated in some, but none required treatment stoppages or side effects. Liver synthetic function was unchanged. Mean 24-h urine copper and zinc excretions at end of treatment were 313.4 ± 191.7 and 2,214 ± 1,346 μg, respectively.
Conclusions
Once-daily trientine should be explored further for possible maintenance therapy for WD. Single daily dose may improve adherence to therapy. Larger trials and longer-term follow-up will establish the safety and treatment efficacy of this once-daily treatment regimen for WD (registration: NCT01472874).
Publisher
Springer US,Springer,Springer Nature B.V
Subject
/ Adult
/ Aged
/ Chelating Agents - administration & dosage
/ Chelating Agents - adverse effects
/ Drug Administration Schedule
/ Female
/ Hepatolenticular Degeneration - diagnosis
/ Hepatolenticular Degeneration - drug therapy
/ Humans
/ Liver
/ Male
/ Medicine
/ Oncology
/ Original
