Fibroblast silver loading for the diagnosis of Menkes disease.

Verheijen, Frans W.; Beerens, Cecile; Havelaar, A.C.; Kleijer, W. J.; Mancini, Grazia M.S.

doi:10.1136/jmg.35.10.849

Cited by 16 publications

(13 citation statements)

References 14 publications

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“…the CPx-type ATPases (based on a conserved intramembranous cysteine-proline-cysteine or cysteine-proline-histidine motif). Direct evidence for a function in copper transport exists only for the CopB ATPase (Enterococcus hirae) [24] and for ATP7A (human), which is defective in Menkes disease [7]. Both proteins can also transport silver.…”

Section: Discussionmentioning

confidence: 99%

“…Recently, we have shown that copper transport can be easily measured using radioactive silver (Ag) [7]. IIHm Ag is commercially available and has a physical half-life of 250 days.…”

Section: Carrier Mediated Uptake Of Iihm Ag In Lysosomal Membranementioning

confidence: 99%

“…Transport studies using radioactive copper are limited by its availability and short physical half-life (12.8 h). Recently, we have shown that radioactive silver can replace copper in copper transport studies [7]. This provides an excellent opportunity to study copper transport mechanisms.…”

Section: Introductionmentioning

confidence: 99%

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Characterization of a heavy metal ion transporter in the lysosomal membrane

et al. 1998

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Lysosomes are thought to play a role in various aspects of heavy metal metabolism. In the present study we demonstrate for the first time the presence of a heavy metal ion transport protein in the lysosomal membrane. Uptake of radioactive silver both in highly purified lysosomal membrane vesicles and in purified intact lysosomes showed the typical kinetics of a carrier-mediated process. This transport was stimulated by ATP hydrolysis, and showed specificity for Ag + , Cu P+ , and Cd P+ . All biochemical properties of this lysosomal metal ion transporter could classify it as a heavy metal transporting P-type ATPase. Long Evans Cinnamon (LEC) rats, an animal model for the copper transport disorder Wilson disease, showed normal lysosomal silver transport.z 1998 Federation of European Biochemical Societies.

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Section: Discussionmentioning

confidence: 99%

“…Recently, we have shown that copper transport can be easily measured using radioactive silver (Ag) [7]. IIHm Ag is commercially available and has a physical half-life of 250 days.…”

Section: Carrier Mediated Uptake Of Iihm Ag In Lysosomal Membranementioning

confidence: 99%

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Characterization of a heavy metal ion transporter in the lysosomal membrane

et al. 1998

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“…Solioz and colleagues [21,22] have used Ag as also a substrate for the bacterial homologue of the Menkes transport protein and it has also been used for the diagnosis of the disease. The approach is limited, however, since the isotope is not commercially available.…”

Section: Discussionmentioning

confidence: 99%

A non-radioactive method for measuring Cu uptake in HepG2 cells

Fosset¹,

McGaw

Reid³

et al. 2005

Journal of Inorganic Biochemistry

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“…Previous studies in bacteria have identified conserved regions of P-type ATPases that are associated with the transport of copper and silver, but not other heavy metals (Solioz and Odermatt, 1995; Stoyanov et al ., 2003). In eukaryotic systems, silver has been shown to utilize the copper-transporter, ATP7A to transport intracellular silver in copper-resistant Chinese hamster ovary (CHO-CUR3) cells and human fibroblasts to the plasma membrane for excretion (Petris et al ., 1996; Verheijen et al ., 1998). ATP7A and the related protein ATP7B are well characterized as copper transporters.…”

Section: Introductionmentioning

confidence: 99%

ATP7B detoxifies silver in ciliated airway epithelial cells

Ibricevic

Brody

Youngs

et al. 2010

Toxicology and Applied Pharmacology

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Silver is a centuries old antibiotic agent currently used to treat infected burns. The sensitivity of a wide range of drug-resistant microorganisms to silver killing suggests that it may be useful for treating refractory lung infections. Toward this goal, we previously developed a methylated caffeine silver acetate compound, SCC1, that exhibits broad-spectrum antimicrobial activity against clinical strains of bacteria in vitro and when nebulized to lungs in mouse infection models. Preclinical testing of high concentrations of SCC1 in primary culture mouse tracheal epithelial cells (mTEC) showed selective ciliated cell death. Ciliated cell death was induced by both silver-and copper-containing compounds, but not by the methylated caffeine portion of SCC1. We hypothesized that copper transporting P-type ATPases, ATP7A and ATP7B, play a role in silver detoxification in the airway. In mTEC, ATP7A was expressed in non-ciliated cells, whereas ATP7B was expressed only in ciliated cells. The exposure of mTEC to SCC1 induced the trafficking of ATP7B, but not ATP7A, suggesting the presence of a cell-specific silver uptake and detoxification mechanisms. Indeed, the expression of the copper uptake protein CTR1 was also restricted to ciliated cells. A role of ATP7B in silver detoxification was further substantiated when treatment of SCC1 significantly increased cell death in ATP7B shRNA treated HepG2 cells. Additionally, mTEC from ATP7B -/-mice showed enhanced loss of ciliated cells compared to wild type. These studies are the first to demonstrate a cell-type specific expression of the Ag + /Cu + transporters ATP7A, ATP7B and CTR1 in airway epithelial cells, and a role for ATP7B in detoxification of these metals in the lung.

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Fibroblast silver loading for the diagnosis of Menkes disease.

Cited by 16 publications

References 14 publications

Characterization of a heavy metal ion transporter in the lysosomal membrane

Characterization of a heavy metal ion transporter in the lysosomal membrane

A non-radioactive method for measuring Cu uptake in HepG2 cells

ATP7B detoxifies silver in ciliated airway epithelial cells

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