Withdrawal of penicillamine from zinc sulphate-penicillamine maintenance therapy in Wilson's disease: promising, safe and cheap

J Neurol Sci. 2008 Jan 15;264(1-2):129-32. doi: 10.1016/j.jns.2007.08.006. Epub 2007 Sep 4.

Abstract

Background: Penicillamine, once considered the cornerstone of treatment for Wilson disease (WD), is rather expensive and toxic, and often causes neurological worsening. Zinc sulphate, aiming at the treatment of free-copper toxicosis, has emerged as effective, safe and cheap alternative.

Aim: To assess the effect of withdrawal of penicillamine from maintenance treatment with penicillamine and zinc sulphate.

Patients and methods: 45 patients of WD (M:F: 28:17; age at diagnosis: 13.5+/-63 years), on both penicillamine (P) and zinc sulphate (Zn), couldn't continue penicillamine due to financial constraints. Their clinical data, disability and impairment scores (Schwab and England (S&E) score, Neurological Symptom Score (NSS), and Chu staging) and follow-up data of patients maintained only on zinc sulphate were recorded.

Results: Majority of patients (84.4%) had neuropsychiatric manifestations. The mean duration of treatment with penicillamine (P) and zinc sulphate (P+Zn), before stopping penicillamine, was 107.4+/-67.3 months. 40 patients improved variably, while the rest didn't. They received only zinc sulphate for 27.2+/-8.5 months (range: 12 to 34) and 44 patients (97.7%) remained status quo or improved marginally. Only one patient reported worsening in dysarthria. Their disability and impairment scores during combination (penicillamine and zinc sulphate) and Zn alone were: Chu (1.3+/-0.5 vs. 1.5+/-1.9; p=0.4), NSS (1.8+/-3.1 vs. 1.5+/-2.3; p=0.03) and S&E (96.4+/-5.6 vs. 98.6+/-3.5; p=0.03). There were no adverse effects.

Conclusions: Withdrawal of penicillamine from zinc sulphate/penicillamine maintenance therapy for patients with Wilson's disease was effective, safe and economic, for almost all patients. This retrospective study reiterates that zinc sulphate may be used as a preferred mode of treatment for patients with Wilson's disease.

MeSH terms

  • Adolescent
  • Adult
  • Astringents / administration & dosage
  • Astringents / economics
  • Chelating Agents / administration & dosage
  • Chelating Agents / adverse effects
  • Chelating Agents / economics
  • Chelation Therapy / adverse effects
  • Chelation Therapy / economics
  • Chelation Therapy / methods
  • Child
  • Child, Preschool
  • Copper / metabolism
  • Copper / toxicity
  • Female
  • Hepatolenticular Degeneration / drug therapy*
  • Hepatolenticular Degeneration / metabolism
  • Hepatolenticular Degeneration / physiopathology
  • Humans
  • Male
  • Neurocognitive Disorders / chemically induced
  • Neurocognitive Disorders / metabolism
  • Neurocognitive Disorders / physiopathology
  • Penicillamine / administration & dosage*
  • Penicillamine / adverse effects
  • Penicillamine / economics
  • Retrospective Studies
  • Treatment Outcome
  • Zinc Sulfate / administration & dosage*
  • Zinc Sulfate / economics

Substances

  • Astringents
  • Chelating Agents
  • Zinc Sulfate
  • Copper
  • Penicillamine