Machine learning suggests polygenic risk for cognitive dysfunction in amyotrophic lateral sclerosis.


Journal

EMBO molecular medicine
ISSN: 1757-4684
Titre abrégé: EMBO Mol Med
Pays: England
ID NLM: 101487380

Informations de publication

Date de publication:
11 01 2021
Historique:
received: 24 04 2020
revised: 27 10 2020
accepted: 30 10 2020
pubmed: 4 12 2020
medline: 6 10 2022
entrez: 3 12 2020
Statut: ppublish

Résumé

Amyotrophic lateral sclerosis (ALS) is a multi-system disease characterized primarily by progressive muscle weakness. Cognitive dysfunction is commonly observed in patients; however, factors influencing risk for cognitive dysfunction remain elusive. Using sparse canonical correlation analysis (sCCA), an unsupervised machine-learning technique, we observed that single nucleotide polymorphisms collectively associate with baseline cognitive performance in a large ALS patient cohort (N = 327) from the multicenter Clinical Research in ALS and Related Disorders for Therapeutic Development (CReATe) Consortium. We demonstrate that a polygenic risk score derived using sCCA relates to longitudinal cognitive decline in the same cohort and also to in vivo cortical thinning in the orbital frontal cortex, anterior cingulate cortex, lateral temporal cortex, premotor cortex, and hippocampus (N = 90) as well as post-mortem motor cortical neuronal loss (N = 87) in independent ALS cohorts from the University of Pennsylvania Integrated Neurodegenerative Disease Biobank. Our findings suggest that common genetic polymorphisms may exert a polygenic contribution to the risk of cortical disease vulnerability and cognitive dysfunction in ALS.

Identifiants

pubmed: 33270986
doi: 10.15252/emmm.202012595
pmc: PMC7799365
doi:

Types de publication

Journal Article Multicenter Study Research Support, N.I.H., Extramural Research Support, Non-U.S. Gov't

Langues

eng

Sous-ensembles de citation

IM

Pagination

e12595

Subventions

Organisme : NIA NIH HHS
ID : R01 AG054060
Pays : United States
Organisme : NIA NIH HHS
ID : P01 AG017586
Pays : United States
Organisme : NINDS NIH HHS
ID : U54 NS092091
Pays : United States
Organisme : NIA NIH HHS
ID : P01 AG066597
Pays : United States
Organisme : NINDS NIH HHS
ID : F31 NS106754
Pays : United States

Informations de copyright

© 2020 The Authors. Published under the terms of the CC BY 4.0 license.

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Auteurs

Katerina Placek (K)

Department of Neurology, University of Pennsylvania Perelman School of Medicine, Philadelphia, PA, USA.

Michael Benatar (M)

Department of Neurology, Leonard M. Miller School of Medicine, University of Miami, Miami, FL, USA.

Joanne Wuu (J)

Department of Neurology, Leonard M. Miller School of Medicine, University of Miami, Miami, FL, USA.

Evadnie Rampersaud (E)

Center for Applied Bioinformatics, St. Jude Children's Research Hospital, Memphis, TN, USA.

Laura Hennessy (L)

Department of Neurology, University of Pennsylvania Perelman School of Medicine, Philadelphia, PA, USA.

Vivianna M Van Deerlin (VM)

Department of Pathology & Laboratory Medicine, University of Pennsylvania Perelman School of Medicine, Philadelphia, PA, USA.

Murray Grossman (M)

Department of Neurology, University of Pennsylvania Perelman School of Medicine, Philadelphia, PA, USA.

David J Irwin (DJ)

Department of Neurology, University of Pennsylvania Perelman School of Medicine, Philadelphia, PA, USA.

Lauren Elman (L)

Department of Neurology, University of Pennsylvania Perelman School of Medicine, Philadelphia, PA, USA.

Leo McCluskey (L)

Department of Neurology, University of Pennsylvania Perelman School of Medicine, Philadelphia, PA, USA.

Colin Quinn (C)

Department of Neurology, University of Pennsylvania Perelman School of Medicine, Philadelphia, PA, USA.

Volkan Granit (V)

Department of Neurology, Leonard M. Miller School of Medicine, University of Miami, Miami, FL, USA.

Jeffrey M Statland (JM)

Department of Neurology, University of Kansas Medical Center, Kansas City, KS, USA.

Ted M Burns (TM)

Department of Neurology, University of Virginia Health System, Charlottesville, VA, USA.

John Ravits (J)

Department of Neurosciences, University of California San Diego, San Diego, CA, USA.

Andrea Swenson (A)

Department of Neurology, University of Iowa, Iowa City, IA, USA.

Jon Katz (J)

Forbes Norris ALS Center, California Pacific Medical Center, San Francisco, CA, USA.

Erik P Pioro (EP)

Department of Neurology, Cleveland Clinic, Cleveland, OH, USA.

Carlayne Jackson (C)

Department of Neurology, University of Texas Health Science Center, San Antonio, TX, USA.

James Caress (J)

Department of Neurology, Wake Forest University School of Medicine, Winston-Salem, NC, USA.

Yuen So (Y)

Department of Neurology, Stanford University Medical Center, San Jose, CA, USA.

Samuel Maiser (S)

Department of Neurology, University of Minnesota Medical Center, Minneapolis, MN, USA.

David Walk (D)

Department of Neurology, University of Minnesota Medical Center, Minneapolis, MN, USA.

Edward B Lee (EB)

Department of Pathology & Laboratory Medicine, University of Pennsylvania Perelman School of Medicine, Philadelphia, PA, USA.

John Q Trojanowski (JQ)

Department of Pathology & Laboratory Medicine, University of Pennsylvania Perelman School of Medicine, Philadelphia, PA, USA.

Philip Cook (P)

Penn Image Computing Science Laboratory (PICSL), Department of Radiology, University of Pennsylvania Perelman School of Medicine, Philadelphia, PA, USA.

James Gee (J)

Penn Image Computing Science Laboratory (PICSL), Department of Radiology, University of Pennsylvania Perelman School of Medicine, Philadelphia, PA, USA.

Jin Sha (J)

Department of Biostatistics, Epidemiology, and Informatics, University of Pennsylvania Perelman School of Medicine, Philadelphia, PA, USA.
Penn Neurodegeneration Genomics Center, Department of Pathology and Laboratory Medicine, University of Pennsylvania Perelman School of Medicine, Philadelphia, PA, USA.

Adam C Naj (AC)

Department of Pathology & Laboratory Medicine, University of Pennsylvania Perelman School of Medicine, Philadelphia, PA, USA.
Department of Biostatistics, Epidemiology, and Informatics, University of Pennsylvania Perelman School of Medicine, Philadelphia, PA, USA.
Penn Neurodegeneration Genomics Center, Department of Pathology and Laboratory Medicine, University of Pennsylvania Perelman School of Medicine, Philadelphia, PA, USA.

Rosa Rademakers (R)

Department of Neuroscience, Mayo Clinic, Jacksonville, FL, USA.
Rare Diseases Clinical Research Network, National Institutes of Health, Bethesda, MD, USA.

Wenan Chen (W)

Center for Applied Bioinformatics, St. Jude Children's Research Hospital, Memphis, TN, USA.

Gang Wu (G)

Center for Applied Bioinformatics, St. Jude Children's Research Hospital, Memphis, TN, USA.

J Paul Taylor (J)

Center for Applied Bioinformatics, St. Jude Children's Research Hospital, Memphis, TN, USA.
The Howard Hughes Medical Institute, Chevy Chase, MS, USA.

Corey T McMillan (CT)

Department of Neurology, University of Pennsylvania Perelman School of Medicine, Philadelphia, PA, USA.

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