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@ARTICLE{Stroo:257883,
author = {Stroo, Esther and Janssen, Leen and Sin, Olga and Hogewerf,
Wytse and Koster, Mirjam and Harkema, Liesbeth and Youssef,
Sameh A and Beschorner, Natalie and Wolters, Anouk Hg and
Bakker, Bjorn and Becker, Lore and Garrett, Lilian and
Marschall, Susan and Hoelter, Sabine M and Wurst, Wolfgang
and Fuchs, Helmut and Gailus-Durner, Valerie and Hrabe de
Angelis, Martin and Thathiah, Amantha and Foijer, Floris and
van de Sluis, Bart and van Deursen, Jan and Jucker, Matthias
and de Bruin, Alain and Nollen, Ellen Aa},
title = {{D}eletion of {SERF}2 in mice delays embryonic development
and alters amyloid deposit structure in the brain.},
journal = {Life science alliance},
volume = {6},
number = {7},
issn = {2575-1077},
address = {Heidelberg},
publisher = {EMBO Press},
reportid = {DZNE-2023-00511},
pages = {e202201730},
year = {2023},
abstract = {In age-related neurodegenerative diseases, like Alzheimer's
and Parkinson's, disease-specific proteins become
aggregation-prone and form amyloid-like deposits. Depletion
of SERF proteins ameliorates this toxic process in worm and
human cell models for diseases. Whether SERF modifies
amyloid pathology in mammalian brain, however, has remained
unknown. Here, we generated conditional Serf2 knockout mice
and found that full-body deletion of Serf2 delayed embryonic
development, causing premature birth and perinatal
lethality. Brain-specific Serf2 knockout mice, on the other
hand, were viable, and showed no major behavioral or
cognitive abnormalities. In a mouse model for amyloid-β
aggregation, brain depletion of Serf2 altered the binding of
structure-specific amyloid dyes, previously used to
distinguish amyloid polymorphisms in the human brain. These
results suggest that Serf2 depletion changed the structure
of amyloid deposits, which was further supported by scanning
transmission electron microscopy, but further study will be
required to confirm this observation. Altogether, our data
reveal the pleiotropic functions of SERF2 in embryonic
development and in the brain and support the existence of
modifying factors of amyloid deposition in mammalian brain,
which offer possibilities for polymorphism-based
interventions.},
keywords = {Brain: embryology / Mice / Humans / Animals / Plaque,
Amyloid: metabolism / Brain: metabolism / Amyloid
beta-Peptides: metabolism / Mice, Knockout / Embryonic
Development: genetics / Mammals: metabolism / Intracellular
Signaling Peptides and Proteins: metabolism / Amyloid
beta-Peptides (NLM Chemicals) / SERF2 protein, human (NLM
Chemicals) / Intracellular Signaling Peptides and Proteins
(NLM Chemicals) / Serf2 protein, mouse (NLM Chemicals)},
cin = {AG Wurst},
ddc = {570},
cid = {I:(DE-2719)1140001},
pnm = {352 - Disease Mechanisms (POF4-352)},
pid = {G:(DE-HGF)POF4-352},
typ = {PUB:(DE-HGF)16},
pubmed = {pmid:37130781},
pmc = {pmc:PMC10155860},
doi = {10.26508/lsa.202201730},
url = {https://pub.dzne.de/record/257883},
}