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Record W4321749735 · doi:10.1093/brain/awad044

Mitochondrial function-associated genes underlie cortical atrophy in prodromal synucleinopathies

2023· article· en· W4321749735 on OpenAlexafffund
Shady Rahayel, Christina Tremblay, Andrew Vo, Bratislav Mišić, Isabelle Arnulf, Jean‐Christophe Corvol, Graziella Mangone, Sara Sambin, Jonas Ihle, Caroline Weill, David Grabli, Florence Cormier‐Dequaire, Louise‐Laure Mariani, Bertrand Degos, Richard J. Levy, Fanny Pineau, Julie Socha, Eve Benchetrit, Virginie Czernecki, Marie-Alexandrine Glachant, Sophie Rivaud-Péchoux, Élodie Hainque, Smaranda Leu Semenescu, Pauline Dodet, Samir Bekadar, Alexis Brice, Suzanne Lesage, Fanny Mochel, Farid Ichou, Vincent Perlbarg, Benoît Colsch, Arthur Tenenhaus, Stéphane Lehéricy, Rahul Gaurav, Nadya Pyatigorskaya, Lydia Yahia‐Cherif, Romain Valabrègue, Cécile Galléa, Marie‐Odile Habert, Dijana Petrovska, Laetitia Jeancolas, Vanessa Brochard, Alizé Chalançon, Carole Dongmo-Kenfack, Christelle Laganot, Valentine Maheo, Jean‐François Gagnon, Ronald B. Postuma, Jacques Montplaisir, Simon J.G. Lewis, Elie Matar, Kaylena A. Ehgoetz Martens, Per Borghammer, Karoline Knudsen, Allan K. Hansen, Oury Monchi, Ziv Gan‐Or, Alain Dagher

Bibliographic record

VenueBrain · 2023
Typearticle
Languageen
FieldMedicine
TopicParkinson's Disease Mechanisms and Treatments
Canadian institutionsUniversity of WaterlooUniversité de MontréalMontreal General HospitalInstitut Universitaire de Gériatrie de MontréalUniversité du Québec à MontréalMcGill UniversityHôpital du Sacré-Cœur de MontréalMontreal Neurological Institute and Hospital
FundersNational Health and Medical Research CouncilNational Institutes of HealthParkinson VerenigingH. Lundbeck A/SJascha FondenÉlectricité de FranceCanadian Institutes of Health ResearchParkinson CanadaLundbeckfondenUniversity of WaterlooAgence Nationale de la RechercheNational Institute on AgingParkinsonforeningenInstitut de FranceMedical Research CouncilBiogenNatural Sciences and Engineering Research Council of CanadaW. Garfield Weston FoundationMichael J. Fox Foundation for Parkinson's Research
KeywordsSynucleinopathiesNeuroscienceAtrophyPosterior cortical atrophyBiologyFunction (biology)MedicineParkinson's diseaseAlpha-synucleinPathologyDementiaDiseaseGenetics

Abstract

fetched live from OpenAlex

Isolated rapid eye movement sleep behaviour disorder (iRBD) is a sleep disorder characterized by the loss of rapid eye movement sleep muscle atonia and the appearance of abnormal movements and vocalizations during rapid eye movement sleep. It is a strong marker of incipient synucleinopathy such as dementia with Lewy bodies and Parkinson's disease. Patients with iRBD already show brain changes that are reminiscent of manifest synucleinopathies including brain atrophy. However, the mechanisms underlying the development of this atrophy remain poorly understood. In this study, we performed cutting-edge imaging transcriptomics and comprehensive spatial mapping analyses in a multicentric cohort of 171 polysomnography-confirmed iRBD patients [67.7 ± 6.6 (49-87) years; 83% men] and 238 healthy controls [66.6 ± 7.9 (41-88) years; 77% men] with T1-weighted MRI to investigate the gene expression and connectivity patterns associated with changes in cortical thickness and surface area in iRBD. Partial least squares regression was performed to identify the gene expression patterns underlying cortical changes in iRBD. Gene set enrichment analysis and virtual histology were then done to assess the biological processes, cellular components, human disease gene terms, and cell types enriched in these gene expression patterns. We then used structural and functional neighbourhood analyses to assess whether the atrophy patterns in iRBD were constrained by the brain's structural and functional connectome. Moreover, we used comprehensive spatial mapping analyses to assess the specific neurotransmitter systems, functional networks, cytoarchitectonic classes, and cognitive brain systems associated with cortical changes in iRBD. All comparisons were tested against null models that preserved spatial autocorrelation between brain regions and compared to Alzheimer's disease to assess the specificity of findings to synucleinopathies. We found that genes involved in mitochondrial function and macroautophagy were the strongest contributors to the cortical thinning occurring in iRBD. Moreover, we demonstrated that cortical thinning was constrained by the brain's structural and functional connectome and that it mapped onto specific networks involved in motor and planning functions. In contrast with cortical thickness, changes in cortical surface area were related to distinct genes, namely genes involved in the inflammatory response, and to different spatial mapping patterns. The gene expression and connectivity patterns associated with iRBD were all distinct from those observed in Alzheimer's disease. In summary, this study demonstrates that the development of brain atrophy in synucleinopathies is constrained by specific genes and networks.

Fetched live from OpenAlex and de-inverted. Abstracts are not stored in this database: the inverted indexes are 8.6 GB of the frame’s 9.3 GB of text, and the host has 13 GB free.

How this classification was reachedexpand

Full frame distilled prediction

Teacher imitation

Not calibrated prevalence, not ground truth. Human validation pending. Learned from the 10,348 direct Codex labels and 10,348 direct Gemma labels. Candidate is the union of thresholded teacher heads; consensus is their intersection. These outputs are machine_predicted_unvalidated and are not human labels or direct frontier model labels.

metaresearch head score (Codex)0.000
metaresearch head score (Gemma)0.000
Version: codex-gemma-dda1882f352aValidation status: machine_predicted_unvalidated
Candidate categoriesnone
Consensus categoriesnone
DomainCandidate signal: none · Consensus signal: none
Study designCandidate signal: Observational · Consensus signal: Observational
GenreCandidate signal: Empirical · Consensus signal: Empirical
Teacher disagreement score0.463
Threshold uncertainty score0.679

Codex and Gemma teacher scores by category

CategoryCodexGemma
Metaresearch0.0000.000
Meta-epidemiology (narrow)0.0000.000
Meta-epidemiology (broad)0.0000.000
Bibliometrics0.0000.000
Science and technology studies0.0000.000
Scholarly communication0.0000.000
Open science0.0000.000
Research integrity0.0000.000
Insufficient payload (model declined to judge)0.0000.001

Machine scores (provisional)

The two teacher heads of the student model, read on this work. A score orders the frame for review; it never asserts a category, and the validation status ships verbatim with every row.

Baseline scores from an immature model (maturity gate not passed, 7 training rounds). Scores rank; they never assert a category.

Opus teacher head0.021
GPT teacher head0.263
Teacher spread0.242 · how far apart the two teachers sit on this one work
Validation statusscore_only:v0-immature-baseline · verbatim from the scoring run: score_only means the number may rank works, and no category label ships from it

Classification

machine, unvalidated

Machine predicted; a candidate call from one teacher head, not a consensus.

The models applied no category: nothing in the taxonomy fit this work.
Study designObservational
Domainnot available
GenreEmpirical

How this classification was reached, model by model and score by score, is at the end of the page under "How this classification was reached".

Quick stats

Citations34
Published2023
Admission routes2
Has abstractyes

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