Literature DB >> 10453198

Treatment and management of Wilson's disease.

N Shimizu1, Y Yamaguchi, T Aoki.   

Abstract

Wilson's disease is an autosomal recessive disorder related to the copper metabolism. The clinical symptoms are due to copper deposition in various tissues, including liver, brain, kidney, cornea and others. The key strategy of treatment is to reduce the amount of copper in the liver and other tissues by administering both copper-chelating agents and a low copper diet. D-Penicillamine is considered to be the first choice as a copper-chelating agent. Patients require 15-25 mg/kg daily in the early stages of treatment and this drug should also be given more than 2 h before meals. Some undesirable or serious side-effects, such as systemic lupus erythematosus (SLE) and nephrotic syndrome, do occur in 20-25% of all patients. In such cases, trienthylene tetramine (trientine) appears to be as effective as penicillamine. This drug is usually used when D-penicillamine has to be withdrawn. It is also sometimes administered to patients with neurological symptoms as a first-choice drug. It is given in doses of 40-50 mg/kg daily, in the same manner as for D-penicillamine. Zinc salt administration has also emerged as an interesting supportive therapy for both treatments. A dose of 5-7.5 mg/kg daily is given before meals. The copper content of the diet should be less than 1 mg/day in the early stages of treatment. Thereafter, it can be increased to 1.0-1.5 mg/day during well-controlled periods. Liver transplantation is now performed in many countries for patients with either the fulminant or chronic progressive types of Wilson's disease.

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Year:  1999        PMID: 10453198     DOI: 10.1046/j.1442-200x.1999.01093.x

Source DB:  PubMed          Journal:  Pediatr Int        ISSN: 1328-8067            Impact factor:   1.524


  5 in total

1.  Co-occurrence of Wilson's disease and systemic lupus erythematosus: a case report and literature review.

Authors:  Lishan Xu; Bin Liu; Zhaoyang Liu; Ning Tang; Chunhui She; Jing Wang; Bo Zang; Yifei Yang
Journal:  BMC Gastroenterol       Date:  2021-05-21       Impact factor: 3.067

2.  Pyridoxal isonicotinoyl hydrazone (PIH) prevents copper-mediated in vitro free radical formation.

Authors:  M Hermes-Lima; M S Gonçalves; R G Andrade
Journal:  Mol Cell Biochem       Date:  2001-12       Impact factor: 3.396

3.  Mutation spectrum of ATP7B gene in pediatric patients with Wilson disease in Vietnam.

Authors:  Nguyen Thi Mai Huong; Nguyen Pham Anh Hoa; Ngo Diem Ngoc; Nguyen Thi Phuong Mai; Pham Hai Yen; Hoàng Thị Vân Anh; Giang Hoa; Tran Minh Dien
Journal:  Mol Genet Metab Rep       Date:  2022-03-15

4.  Surface-engineered nanoliposomes by chelating ligands for modulating the neurotoxicity associated with β-amyloid aggregates of Alzheimer's disease.

Authors:  Maluta S Mufamadi; Yahya E Choonara; Pradeep Kumar; Girish Modi; Dinesh Naidoo; Valence M K Ndesendo; Lisa C du Toit; Sunny E Iyuke; Viness Pillay
Journal:  Pharm Res       Date:  2012-05-15       Impact factor: 4.200

Review 5.  Canine Models for Copper Homeostasis Disorders.

Authors:  Xiaoyan Wu; Peter A J Leegwater; Hille Fieten
Journal:  Int J Mol Sci       Date:  2016-02-04       Impact factor: 5.923

  5 in total

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