Abstract

The function of human Sco1 and Sco2 is shown to be dependent on copper ion binding. Expression of soluble domains of human Sco1 and Sco2 either in bacteria or the yeast cytoplasm resulted in the recovery of copper-containing proteins. The metallation of human Sco1, but not Sco2, when expressed in the yeast cytoplasm is dependent on the co-expression of human Cox17. Two conserved cysteines and a histidyl residue, known to be important for both copper binding and in vivo function in yeast Sco1, are also critical for in vivo function of human Sco1 and Sco2. Human and yeast Sco proteins can bind either a single Cu(I) or Cu(II) ion. The Cu(II) site yields S-Cu(II) charge transfer transitions that are not bleached by weak reductants or chelators. The Cu(I) site exhibits trigonal geometry, whereas the Cu(II) site resembles a type II Cu(II) site with a higher coordination number. To identify additional potential ligands for the Cu(II) site, a series of mutant proteins with substitutions in conserved residues in the vicinity of the Cu(I) site were examined. Mutation of several conserved carboxylates did not alter either in vivo function or the presence of the Cu(II) chromophore. In contrast, replacement of Asp238 in human or yeast Sco1 abrogated the Cu(II) visible transitions and in yeast Sco1 attenuated Cu(II), but not Cu(I), binding. Both the mutant yeast and human proteins were nonfunctional, suggesting the importance of this aspartate for normal function. Taken together, these data suggest that both Cu(I) and Cu(II) binding are critical for normal Sco function.

Highlights

  • In membrane insertion of subunits, and in either the synthesis or delivery of cofactors

  • This study firmly establishes that both human Sco1 and Sco2 are copper-binding proteins and that the function of each is dependent on copper ion binding

  • Four lines of evidence support specific copper ion binding to human Sco1 and Sco2

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Summary

Introduction

In membrane insertion of subunits, and in either the synthesis or delivery of cofactors. Sco and Cox are inner membrane proteins tethered by a single transmembrane helix and are implicated in the assembly of the CuA and CuB centers, respectively [6, 7]. Both proteins are copper-binding proteins, and mutations that abrogate Cu(I) binding attenuate in vivo assembly of CcO (8 –11). The CcO deficiency in SCO1 and SCO2 patient cell lines is partially suppressed by the addition of exogenous copper to the culture medium [18, 21, 22] It is unclear why mammalian cells require two distinct Sco molecules for CcO maturation. The distinctive clinical presentation is not a result of tissue-specific expression of the two genes, since SCO1 and SCO2 are ubiquitously expressed and exhibit a similar expression pattern in dif-

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