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@ARTICLE{Aldobiyan:642771,
author = {Aldobiyan, Ibrahim and Elliston, Emma L. K. and
Heyer-Chauhan, Narinder and Arold, Stefan T. and Zhao,
Lingyun and Huntington, Brandon and Lowen, Sarah M. and
Orlova, Elena V. and Irving, James and Lomas, David A.},
title = {{T}he mechanism of pathogenic α 1 -antitrypsin aggregation
in the human liver},
journal = {Proceedings of the National Academy of Sciences of the
United States of America},
volume = {122},
number = {46},
issn = {0027-8424},
address = {Washington, DC},
publisher = {National Acad. of Sciences},
reportid = {PUBDB-2025-05620},
pages = {e2507535122},
year = {2025},
abstract = {Originating 2 to 3 millennia ago in a Scandinavian
population, the SERPINA1 Z allele (Glu342Lys) is present in
up to $2.5\%$ of populations of Northern European descent
and accounts for $95\%$ of severe α1-antitrypsin
deficiency. The α1-antitrypsin Z variant self-assembles
into polymer chains that deposit within hepatocytes,
predisposing to liver disease. Here, the 4.0Å subunit
structure of polymers isolated directly from human liver
tissue has been determined using cryoelectron microscopy.
Challenges of flexibility, small subunit size, heterogeneous
length, and preferred orientations were mitigated using
antibody Fab domains and sample preparation strategies. This
structure demonstrates that the formation of polymers in
vivo involves self-incorporation of an exposed structural
element (the reactive center loop) as an additional
β-strand into the central β-sheet of α1-antitrypsin and
displacement of a C-terminal region from one subunit with
incorporation into the next. Unlike amyloid aggregation,
this well-folded structure partially recapitulates a
conformation adopted during normal function of the protein.
These perturbations to the constituent α1-antitrypsin
subunits of human tissue-derived polymers are consistent
with a pronounced stability, their tendency toward
long-chain forms, the ability of a subset to undergo
canonical secretion, and the action of a class of small
molecules that block polymerization in vivo.},
cin = {EMBL-User},
ddc = {500},
cid = {I:(DE-H253)EMBL-User-20120814},
pnm = {6G3 - PETRA III (DESY) (POF4-6G3)},
pid = {G:(DE-HGF)POF4-6G3},
experiment = {EXP:(DE-H253)P-P14-20150101},
typ = {PUB:(DE-HGF)16},
doi = {10.1073/pnas.2507535122},
url = {https://bib-pubdb1.desy.de/record/642771},
}