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@ARTICLE{Meyer:904805,
author = {Meyer, Magdalena and Jurek, Benjamin and Alfonso-Prieto,
Mercedes and Ribeiro, Rui and Milenkovic, Vladimir M. and
Winter, Julia and Hoffmann, Petra and Wetzel, Christian H.
and Giorgetti, Alejandro and Carloni, Paolo and Neumann,
Inga D.},
title = {{S}tructure-function relationships of the disease-linked
{A}218{T} oxytocin receptor variant},
journal = {Molecular psychiatry},
volume = {27},
issn = {1359-4184},
address = {London},
publisher = {Macmillan},
reportid = {FZJ-2022-00135},
pages = {907–917},
year = {2022},
note = {Open Access Article. Open Access funding enabled and
organized by Projekt DEAL through University of Regensburg.},
abstract = {Various single nucleotide polymorphisms (SNPs) in the
oxytocin receptor (OXTR) gene have been associated with
behavioral traits, autism spectrum disorder (ASD) and other
diseases. The non-synonymous SNP rs4686302 results in the
OXTR variant A218T and has been linked to core
characteristics of ASD, trait empathy and preterm birth.
However, the molecular and intracellular mechanisms
underlying those associations are still elusive. Here, we
uncovered the molecular and intracellular consequences of
this mutation that may affect the psychological or
behavioral outcome of oxytocin (OXT)-treatment regimens in
clinical studies, and provide a mechanistic explanation for
an altered receptor function. We created two monoclonal
HEK293 cell lines, stably expressing either the wild-type or
A218T OXTR. We detected an increased OXTR protein stability,
accompanied by a shift in Ca2+ dynamics and reduced MAPK
pathway activation in the A218T cells. Combined whole-genome
and RNA sequencing analyses in OXT-treated cells revealed
7823 differentially regulated genes in A218T compared to
wild-type cells, including 429 genes being associated with
ASD. Furthermore, computational modeling provided a
molecular basis for the observed change in OXTR stability
suggesting that the OXTR mutation affects downstream events
by altering receptor activation and signaling, in agreement
with our in vitro results. In summary, our study provides
the cellular mechanism that links the OXTR rs4686302 SNP
with genetic dysregulations associated with aspects of ASD.},
cin = {IAS-5 / INM-9},
ddc = {610},
cid = {I:(DE-Juel1)IAS-5-20120330 / I:(DE-Juel1)INM-9-20140121},
pnm = {5241 - Molecular Information Processing in Cellular Systems
(POF4-524) / 5251 - Multilevel Brain Organization and
Variability (POF4-525) / 5252 - Brain Dysfunction and
Plasticity (POF4-525)},
pid = {G:(DE-HGF)POF4-5241 / G:(DE-HGF)POF4-5251 /
G:(DE-HGF)POF4-5252},
typ = {PUB:(DE-HGF)16},
pubmed = {pmid:34980886},
UT = {WOS:000737750600001},
doi = {10.1038/s41380-021-01241-8},
url = {https://juser.fz-juelich.de/record/904805},
}