Literature DB >> 16098018

Copper-replacement treatment for symptomatic Menkes disease: ethical considerations.

S R Sheela1, M Latha, P Liu, K Lem, S G Kaler.   

Abstract

We describe a child with classical Menkes disease with a novel ATP7A mutation, intractable seizures, severe hypotonia and developmental delay, hypopigmentation of the skin and hair, and failure to thrive, who was treated with daily subcutaneous copper histidine injections for 2(1/2) years, beginning at 15 months of age. He became seizure-free and pigmentation of his skin and hair darkened, but he continued to have severe developmental delays. His condition remains stable 8 months after stopping treatment. We review the ethical aspects of offering copper treatment for Menkes disease infants diagnosed after neurological symptoms become manifest. These include (1) the prospect for any benefits, (2) the potential risks and discomforts, (3) the parents' wishes with respect to treatment, (4) the family's understanding of the treatment's potential futility, (5) the family's understanding of the investigational nature of this treatment, (6) the potential for treatment to have an adverse impact on unaffected family members, (7) whether the ultimate decision regarding treatment should rest with health care providers or with the patient's parents, and (8) the duration of treatment. The ethical issues encountered in providing possibly futile treatment in this difficult disorder seem relevant to other pediatric medical conditions as well.

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Year:  2005        PMID: 16098018     DOI: 10.1111/j.1399-0004.2005.00496.x

Source DB:  PubMed          Journal:  Clin Genet        ISSN: 0009-9163            Impact factor:   4.438


  18 in total

Review 1.  Research challenges in central nervous system manifestations of inborn errors of metabolism.

Authors:  P I Dickson; A R Pariser; S C Groft; R W Ishihara; D E McNeil; D Tagle; D J Griebel; S G Kaler; J W Mink; E G Shapiro; K J Bjoraker; L Krivitzky; J M Provenzale; A Gropman; P Orchard; G Raymond; B H Cohen; R D Steiner; S F Goldkind; R M Nelson; E Kakkis; M C Patterson
Journal:  Mol Genet Metab       Date:  2010-12-02       Impact factor: 4.797

Review 2.  Peptidylgycine α-amidating monooxygenase and copper: a gene-nutrient interaction critical to nervous system function.

Authors:  Danielle Bousquet-Moore; Richard E Mains; Betty A Eipper
Journal:  J Neurosci Res       Date:  2010-09       Impact factor: 4.164

Review 3.  ATP7A-related copper transport diseases-emerging concepts and future trends.

Authors:  Stephen G Kaler
Journal:  Nat Rev Neurol       Date:  2011-01       Impact factor: 42.937

4.  Tandem Duplication of Exons 1-7 Neither Impairs ATP7A Expression Nor Causes a Menkes Disease Phenotype.

Authors:  Eun-Young Choi; Keyur Patel; Marie Reine Haddad; Ling Yi; Courtney Holmes; David S Goldstein; Amalia Dutra; Evgenia Pak; Stephen G Kaler
Journal:  JIMD Rep       Date:  2015-02-01

5.  Cervical spine anomalies in Menkes disease: a radiologic finding potentially confused with child abuse.

Authors:  Suvimol C Hill; Andrew J Dwyer; Stephen G Kaler
Journal:  Pediatr Radiol       Date:  2012-07-24

Review 6.  Inborn errors of copper metabolism.

Authors:  Stephen G Kaler
Journal:  Handb Clin Neurol       Date:  2013

Review 7.  Catecholamine metabolites affected by the copper-dependent enzyme dopamine-beta-hydroxylase provide sensitive biomarkers for early diagnosis of menkes disease and viral-mediated ATP7A gene therapy.

Authors:  Stephen G Kaler; Courtney S Holmes
Journal:  Adv Pharmacol       Date:  2013

Review 8.  Molecular pathogenesis of Wilson and Menkes disease: correlation of mutations with molecular defects and disease phenotypes.

Authors:  P de Bie; P Muller; C Wijmenga; L W J Klomp
Journal:  J Med Genet       Date:  2007-08-23       Impact factor: 6.318

9.  Interactions of peptide amidation and copper: novel biomarkers and mechanisms of neural dysfunction.

Authors:  Danielle Bousquet-Moore; Joseph R Prohaska; Eduardo A Nillni; Traci Czyzyk; William C Wetsel; Richard E Mains; Betty A Eipper
Journal:  Neurobiol Dis       Date:  2009-10-06       Impact factor: 5.996

10.  In vivo correction of a Menkes disease model using antisense oligonucleotides.

Authors:  Erik C Madsen; Paul A Morcos; Bryce A Mendelsohn; Jonathan D Gitlin
Journal:  Proc Natl Acad Sci U S A       Date:  2008-03-03       Impact factor: 11.205

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