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Prospective pilot study of a single daily dosage of trientine for the treatment of Wilson disease.

AbstractBACKGROUND:
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).
AuthorsAftab Ala, Ermal Aliu, Michael L Schilsky
JournalDigestive diseases and sciences (Dig Dis Sci) Vol. 60 Issue 5 Pg. 1433-9 (May 2015) ISSN: 1573-2568 [Electronic] United States
PMID25605552 (Publication Type: Clinical Trial, Journal Article, Research Support, Non-U.S. Gov't)
Chemical References
  • Chelating Agents
  • Trientine
Topics
  • Administration, Oral
  • Adult
  • Aged
  • Chelating Agents (administration & dosage, adverse effects)
  • Drug Administration Schedule
  • Female
  • Hepatolenticular Degeneration (diagnosis, drug therapy)
  • Humans
  • Male
  • Medication Adherence
  • Middle Aged
  • Patient Dropouts
  • Patient Satisfaction
  • Pilot Projects
  • Prospective Studies
  • Surveys and Questionnaires
  • Time Factors
  • Treatment Outcome
  • Trientine (administration & dosage, adverse effects)
  • Young Adult

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