Evidence map›Paper›PMID 37247383›Full record

ArticleBrain : a journal of neurology2023

Lysosomal polygenic risk is associated with the severity of neuropathology in Lewy body disease.

Jon-Anders Tunold, Manuela M X Tan, Shunsuke Koga, Hanneke Geut, Annemieke J M Rozemuller, Rebecca Valentino, Hiroaki Sekiya, Nicholas B Martin, Michael G Heckman, Jose Bras and 6 more

Open access · hybridAbstract read
In one paragraph

Article in Brain : a journal of neurology, 2023. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 15 papers.

0numbers the graph read from it
0cells of the map it votes in
15citing papers in PubMed
5.6field-weighted citation impact, top 3% of its field
1 · What the graph read from it

What it found

Each row is one number read from the abstract, on the scale the paper reported it, with its interval. Left of the dashed line favours the treatment, right favours the comparator. Under each row is the sentence it came from. New to these charts? A ten-minute tutorial.

The abstract states no effect estimate the extractor could read, or names no intervention and outcome on the map, so this paper lights no cell and moves no belief. It is still indexed, cited and linked below.

2 · The registry

The trial behind it

Trials whose registry record cites this paper, or whose number appears in the abstract. A trial that started after this paper was published is citing it as background, not reporting it.

Neither the registry nor the abstract names a trial number. If this is a trial report, that itself is worth knowing.

3 · Its place in the literature

Who cites it

15 citing papers in PubMed, 27 citations in OpenAlex.

  1. Rare-Variant Burden across Lysosomal Genes Implicates Sialylation and Ganglioside Metabolism in Parkinson's Disease.Movement disorders : official journal of the Movement Disorder Society · 2026
    Article
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  4. Review
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  8. Article
  9. Levodopa-induced dyskinesia: brain iron deposition as a new hypothesis.Biometals : an international journal on the role of metal ions in biology, biochemistry, and medicine · 2024
    Review
  10. Article
  11. Article
  12. Refining the clinical diagnosis of Parkinson's disease.Parkinsonism & related disorders · 2024
    Review
  13. Frequency of Comorbid Pathologies and Their Clinical Impact in Multiple System Atrophy.Movement disorders : official journal of the Movement Disorder Society · 2024
    Article
  14. Review
  15. Article
4 · The record

Corrections and comments

PubMed lists nothing against this paper. Absence here is not a guarantee, only a check that was made.

5 · Who and what money

Authors and funding

16 authors at 5 institutions in 3 countries.

Jon-Anders TunoldDepartment of Neurology, Oslo University Hospital, 0424 Oslo, Norway.
Manuela M X TanDepartment of Neurology, Oslo University Hospital, 0424 Oslo, Norway.
Shunsuke KogaDepartment of Neuroscience, Mayo Clinic, Jacksonville, FL 32224, USA.ORCID 0000-0001-8868-9700
Hanneke GeutDepartment of Anatomy and Neurosciences, Amsterdam UMC, Vrije Universiteit, 1081 HV Amsterdam, The Netherlands.
Annemieke J M RozemullerDepartment of Anatomy and Neurosciences, Amsterdam UMC, Vrije Universiteit, 1081 HV Amsterdam, The Netherlands.
Rebecca ValentinoDepartment of Neuroscience, Mayo Clinic, Jacksonville, FL 32224, USA.
Hiroaki SekiyaDepartment of Neuroscience, Mayo Clinic, Jacksonville, FL 32224, USA.
Nicholas B MartinDepartment of Neuroscience, Mayo Clinic, Jacksonville, FL 32224, USA.
Michael G HeckmanDivision of Clinical Trials and Biostatistics, Mayo Clinic, Jacksonville, FL 32224, USA.
Jose BrasDepartment of Neurodegenerative Science, Van Andel Institute, Grand Rapids, MI 49503, USA.
Rita GuerreiroDepartment of Neurodegenerative Science, Van Andel Institute, Grand Rapids, MI 49503, USA.
Dennis W DicksonDepartment of Neuroscience, Mayo Clinic, Jacksonville, FL 32224, USA.ORCID 0000-0001-7189-7917
Mathias ToftDepartment of Neurology, Oslo University Hospital, 0424 Oslo, Norway.
Wilma D J van de BergDepartment of Anatomy and Neurosciences, Amsterdam UMC, Vrije Universiteit, 1081 HV Amsterdam, The Netherlands.
Owen A RossDepartment of Neuroscience, Mayo Clinic, Jacksonville, FL 32224, USA.
Lasse PihlstrømDepartment of Neurology, Oslo University Hospital, 0424 Oslo, Norway.ORCID 0000-0002-7635-8645
Mayo Clinic in Florida · USOslo University Hospital · NOAmsterdam Neuroscience · NLVan Andel Institute · USAmsterdam University Medical Centers · NL

Funding

Vascular Structure and Function in Cognitive AgingP01AG003949 · NIA · YESHIVA UNIVERSITY · PI Charles B Hall · 1985 to 2026
$73.9M
Project: Predicting PhenoconversionU19AG071754 · NIA · WASHINGTON UNIVERSITY · PI Bradley F Boeve, Yo-El S Ju · 2021 to 2026
$41.2M
Longitudinal Imaging Biomarkers of Prodromal DLBU01NS100620 · NINDS · MAYO CLINIC ROCHESTER · PI Bradley F Boeve, KEJAL KANTARCI · 2017 to 2026
$19.2M
Utilization of proteomics and lipidomics to identify modifiers of LBDU54NS110435 · NINDS · MAYO CLINIC JACKSONVILLE · PI MCLEAN, PAMELA J · 2019 to 2023
$14.5M
Investigating Resistance and Resilience Mechanisms in Alzheimer’s DiseaseR01AG056366 · NIA · MAYO CLINIC ROCHESTER · PI PRASHANTHI VEMURI · 2017 to 2026
$7.0M
Project 2: Identifying genes and Pathways that impact Tau Toxicity in FTDU54NS100693 · NINDS · MAYO CLINIC JACKSONVILLE · PI PETRUCELLI, LEONARD · 2016 to 2020
$6.2M
Molecular mechanisms of PINK1-PRKN directed mitochondrial quality controlRF1NS085070 · NINDS · MAYO CLINIC JACKSONVILLE · PI SPRINGER, WOLFDIETER · 2019 to 2019
$2.8M
NIA NIH HHS P01 AG003949NIA NIH HHS R01 AG056366NIA NIH HHS U19 AG071754NINDS NIH HHS RF1 NS085070NINDS NIH HHS U01 NS100620NINDS NIH HHS U54 NS100693NINDS NIH HHS U54 NS110435
6 · The paper itself

Abstract

Intraneuronal accumulation of misfolded α-synuclein is the pathological hallmark of Parkinson's disease and dementia with Lewy bodies, often co-occurring with variable degrees of Alzheimer's disease related neuropathology. Genetic association studies have successfully identified common variants associated with disease risk and phenotypic traits in Lewy body disease, yet little is known about the genetic contribution to neuropathological heterogeneity. Using summary statistics from Parkinson's disease and Alzheimer's disease genome-wide association studies, we calculated polygenic risk scores and investigated the relationship with Lewy, amyloid-β and tau pathology. Associations were nominated in neuropathologically defined samples with Lewy body disease from the Netherlands Brain Bank (n = 217) and followed up in an independent sample series from the Mayo Clinic Brain Bank (n = 394). We also generated stratified polygenic risk scores based on single-nucleotide polymorphisms annotated to eight functional pathways or cell types previously implicated in Parkinson's disease and assessed for association with Lewy pathology in subgroups with and without significant Alzheimer's disease co-pathology. In an ordinal logistic regression model, the Alzheimer's disease polygenic risk score was associated with concomitant amyloid-β and tau pathology in both cohorts. Moreover, both cohorts showed a significant association between lysosomal pathway polygenic risk and Lewy pathology, which was more consistent than the association with a general Parkinson's disease risk score and specific to the subset of samples without significant concomitant Alzheimer's disease related neuropathology. Our findings provide proof of principle that the specific risk alleles a patient carries for Parkinson's and Alzheimer's disease also influence key aspects of the underlying neuropathology in Lewy body disease. The interrelations between genetic architecture and neuropathology are complex, as our results implicate lysosomal risk loci specifically in the subset of samples without Alzheimer's disease co-pathology. Our findings hold promise that genetic profiling may help predict the vulnerability to specific neuropathologies in Lewy body disease, with potential relevance for the further development of precision medicine in these disorders.

Indexed as

Alzheimer DiseaseLewy Body DiseaseParkinson DiseaseAmyloid beta-PeptidesGenome-Wide Association StudyHumansLysosomesAmyloid beta-PeptidesgeneticsLewy body diseaselysosomal pathwayneuropathologyParkinson’s disease

Identifiers

PMID37247383
PMCPMC10545498
OpenAlexW4378647721

What OpenQuestion holds

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LicenceCC BY-NC
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Registered trials

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Read under generation 80e0d062 · epoch 390. Bibliography from PubMed, PubMed Central and OpenAlex; grants from NIH RePORTER; trial links from ClinicalTrials.gov; estimates, votes and beliefs from the OpenQuestion graph.