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Referenced in 2 patents
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Research Article Free access | 10.1172/JCI119397

Scott syndrome erythrocytes contain a membrane protein capable of mediating Ca2+-dependent transbilayer migration of membrane phospholipids.

J G Stout, F Bassé, R A Luhm, H J Weiss, T Wiedmer, and P J Sims

Blood Research Institute of The Blood Center of Southeastern Wisconsin, Milwaukee 53201, USA.

Find articles by Stout, J. in: PubMed | Google Scholar

Blood Research Institute of The Blood Center of Southeastern Wisconsin, Milwaukee 53201, USA.

Find articles by Bassé, F. in: PubMed | Google Scholar

Blood Research Institute of The Blood Center of Southeastern Wisconsin, Milwaukee 53201, USA.

Find articles by Luhm, R. in: PubMed | Google Scholar

Blood Research Institute of The Blood Center of Southeastern Wisconsin, Milwaukee 53201, USA.

Find articles by Weiss, H. in: PubMed | Google Scholar

Blood Research Institute of The Blood Center of Southeastern Wisconsin, Milwaukee 53201, USA.

Find articles by Wiedmer, T. in: PubMed | Google Scholar

Blood Research Institute of The Blood Center of Southeastern Wisconsin, Milwaukee 53201, USA.

Find articles by Sims, P. in: PubMed | Google Scholar

Published May 1, 1997 - More info

Published in Volume 99, Issue 9 on May 1, 1997
J Clin Invest. 1997;99(9):2232–2238. https://doi.org/10.1172/JCI119397.
© 1997 The American Society for Clinical Investigation
Published May 1, 1997 - Version history
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Abstract

Phospholipid (PL) scramblase is a plasma membrane protein that mediates accelerated transbilayer migration of PLs upon binding Ca2+, facilitating rapid mobilization of phosphatidylserine to the cell surface upon elevation of internal Ca2+. In patients with Scott syndrome, a congenital bleeding disorder related to defective expression of membrane coagulant activity, circulating blood cells show decreased cell surface exposure of phosphatidylserine at elevated cytosolic [Ca2+], implying an underlying defect or deficiency of PL scramblase. To gain insight into the molecular basis of this disorder, we compared PL scramblase in Scott erythrocyte membranes to those of normal controls. Whereas membranes of Scott cells were unresponsive to Ca2+-induced activation of PL scramblase at neutral pH, apparently normal PL scramblase activity was induced at pH < 6.0. After extraction with octylglucoside, a membrane protein was isolated from the Scott cells which exhibited normal PL scramblase activity when reconstituted in vesicles with exogenous PLs. Like PL scramblase from normal erythrocytes, PL scramblase from Scott erythrocytes was maximally activated either by addition of Ca2+ (at pH 7.4) or by acidification to pH < 6.0, and similar apparent affinities for Ca2+ and rates of transbilayer transfer of PLs were observed. This suggests that the defect in Scott syndrome is related to an altered interaction of Ca2+ with PL scramblase on the endofacial surface of the cell membrane, due either to an intrinsic constraint upon the protein preventing interaction with Ca2+ in situ, or due to an unidentified inhibitor or cofactor in the Scott cell that is dissociated by detergent.

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Referenced in 2 patents
19 readers on Mendeley
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