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@ARTICLE{Heger:285208,
author = {Heger, Leonie and Gubinelli, Francesco and Huber, Andreas J
and Cardona-Alberich, Aida and Rovere, Matteo and Matti, Ulf
and Müller, Stephan A and Nagaraja, Sankarshana and
Jaschkowitz, Lena and Schifferer, Martina and Wurst,
Wolfgang and Lichtenthaler, Stefan F and Behrends, Christian
and Sambandan, Sivakumar and Burbulla, Lena F.},
title = {{VMAT}2 dysfunction impairs vesicular dopamine uptake,
driving its oxidation and α-synuclein pathology in
{DJ}-1-linked {P}arkinson's neurons.},
journal = {Science advances},
volume = {12},
number = {7},
issn = {2375-2548},
address = {Washington, DC [u.a.]},
publisher = {Assoc.},
reportid = {DZNE-2026-00187},
pages = {eadz5645},
year = {2026},
abstract = {Parkinson's disease (PD) is characterized by α-synuclein
accumulation and dopaminergic neuron degeneration, with
dopamine (DA) oxidation emerging as a key pathological
driver. However, the mechanisms underlying this neurotoxic
process remain unclear. Using PD patient-derived and
CRISPR-engineered induced pluripotent stem cell midbrain
dopaminergic neurons lacking DJ-1, we identified defective
sequestration of cytosolic DA into synaptic vesicles, which
culminated in DA oxidation and α-synuclein pathology.
In-depth proteomics, state-of-the-art imaging, and
ultrasensitive DA probes uncovered that decreased vesicular
monoamine transporter 2 (VMAT2) protein and function
impaired vesicular DA uptake, resulting in reduced vesicle
availability and abnormal vesicle morphology. Furthermore,
VMAT2 activity and vesicle endocytosis are processes
dependent on adenosine 5'-triphosphate (ATP), which is
notably reduced in DJ-1-deficient dopaminergic neurons. ATP
supplementation restored vesicular function and alleviated
DA-related pathologies in mutant dopaminergic neurons. This
study reveals an ATP-sensitive mechanism that regulates DA
homeostasis through VMAT2 and vesicle dynamics in midbrain
dopaminergic neurons, highlighting enhanced DA sequestration
as a promising therapeutic strategy for PD.},
keywords = {Vesicular Monoamine Transport Proteins: metabolism /
Vesicular Monoamine Transport Proteins: genetics / Dopamine:
metabolism / Parkinson Disease: metabolism / Parkinson
Disease: pathology / Parkinson Disease: genetics / Protein
Deglycase DJ-1: genetics / Protein Deglycase DJ-1:
metabolism / Humans / Dopaminergic Neurons: metabolism /
Dopaminergic Neurons: pathology / alpha-Synuclein:
metabolism / alpha-Synuclein: genetics / Oxidation-Reduction
/ Animals / Synaptic Vesicles: metabolism / Mice / Adenosine
Triphosphate: metabolism / Induced Pluripotent Stem Cells:
metabolism / Mesencephalon: metabolism / Vesicular Monoamine
Transport Proteins (NLM Chemicals) / Dopamine (NLM
Chemicals) / Protein Deglycase DJ-1 (NLM Chemicals) /
alpha-Synuclein (NLM Chemicals) / SLC18A2 protein, human
(NLM Chemicals) / Adenosine Triphosphate (NLM Chemicals) /
PARK7 protein, human (NLM Chemicals)},
cin = {AG Burbulla / AG Lichtenthaler / AG Misgeld / AG Wurst},
ddc = {500},
cid = {I:(DE-2719)5000074 / I:(DE-2719)1110006 /
I:(DE-2719)1110000-4 / I:(DE-2719)1140001},
pnm = {352 - Disease Mechanisms (POF4-352) / 351 - Brain Function
(POF4-351)},
pid = {G:(DE-HGF)POF4-352 / G:(DE-HGF)POF4-351},
typ = {PUB:(DE-HGF)16},
pubmed = {pmid:41671379},
pmc = {pmc:PMC12893317},
doi = {10.1126/sciadv.adz5645},
url = {https://pub.dzne.de/record/285208},
}