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@ARTICLE{Oppici:166796,
author = {Oppici, Elisa and Fodor, Krisztian and Paiardini,
Alessandro and Williams, Chris and Voltattorni, Carla Borri
and Wilmanns, Matthias and Cellini, Barbara},
title = {{C}rystal structure of the {S}187{F} variant of human liver
alanine: {A}minotransferase associated with primary
hyperoxaluria type {I} and its functional implications},
journal = {Proteins},
volume = {81},
number = {8},
issn = {0887-3585},
address = {New York, NY},
publisher = {Wiley-Liss},
reportid = {DESY-2014-01630},
pages = {1457 - 1465},
year = {2013},
note = {(c) WILEY PERIODICALS, INC},
abstract = {The substitution of Ser187, a residue located far from the
active site of human liver peroxisomal alanine:glyoxylate
aminotransferase (AGT), by Phe gives rise to a variant
associated with primary hyperoxaluria type I. Unexpectedly,
previous studies revealed that the recombinant form of S187F
exhibits a remarkable loss of catalytic activity, an
increased pyridoxal 5'-phosphate (PLP) binding affinity and
a different coenzyme binding mode compared with normal AGT.
To shed light on the structural elements responsible for
these defects, we solved the crystal structure of the
variant to a resolution of 2.9 Å. Although the overall
conformation of the variant is similar to that of normal
AGT, we noticed: (i) a displacement of the PLP-binding
Lys209 and Val185, located on the re and si side of PLP,
respectively, and (ii) slight conformational changes of
other active site residues, in particular Trp108, the base
stacking residue with the pyridine cofactor moiety. This
active site perturbation results in a mispositioning of the
AGT-pyridoxamine 5'-phosphate (PMP) complex and of the
external aldimine, as predicted by molecular modeling
studies. Taken together, both predicted and observed
movements caused by the S187F mutation are consistent with
the following functional properties of the variant: (i) a
300- to 500-fold decrease in both the rate constant of
L-alanine half-transamination and the kcat of the overall
transamination, (ii) a different PMP binding mode and
affinity, and (iii) a different microenvironment of the
external aldimine. Proposals for the treatment of patients
bearing S187F mutation are discussed on the basis of these
results.},
cin = {EMBL},
ddc = {540},
cid = {I:(DE-H253)EMBL-20120731},
pnm = {DORIS Beamline K1.3 (POF2-54G13)},
pid = {G:(DE-H253)POF2-K1.3-20130405},
experiment = {EXP:(DE-H253)D-K1.3-20150101},
typ = {PUB:(DE-HGF)16},
UT = {WOS:000329220400015},
pubmed = {pmid:23589421},
doi = {10.1002/prot.24300},
url = {https://bib-pubdb1.desy.de/record/166796},
}