Cited by 117Amyotrophic Lateral Sclerosis ResearchRNA modifications and cancerAlzheimer's disease research and treatments

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RNA methylation influences TDP43 binding and disease pathogenesis in models of amyotrophic lateral sclerosis and frontotemporal dementia

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McMillan, Michael, Gomez, Nicolas, Hsieh, Caroline, Bekier, Michael, Li, Xingli, Miguez, Roberto, Tank, Elizabeth M.H., Barmada, Sami J.·2023·Molecular Cell

DOI 10.1016/j.molcel.2022.12.019PMC PMC9899051

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Abstract

RNA methylation at adenosine N6 (m6A) is one of the most common RNA modifications, impacting RNA stability, transport, and translation. Previous studies uncovered RNA destabilization in amyotrophic lateral sclerosis (ALS) models in association with accumulation of the RNA-binding protein TDP43. Here, we show that TDP43 recognizes m6A RNA and that RNA methylation is critical for both TDP43 binding and autoregulation. We also observed extensive RNA hypermethylation in ALS spinal cord, corresponding to methylated TDP43 substrates. Emphasizing the importance of m6A for TDP43 binding and function, we identified several m6A factors that enhance or suppress TDP43-mediated toxicity via single-cell CRISPR-Cas9 in primary neurons. The most promising modifier-the canonical m6A reader YTHDF2-accumulated within ALS spinal neurons, and its knockdown prolonged the survival of human neurons carrying ALS-associated mutations. Collectively, these data show that m6A modifications modulate RNA binding by TDP43 and that m6A is pivotal for TDP43-related neurodegeneration in ALS.

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Authors: McMillan, M., Gomez, N., Hsieh, C., Bekier, M., Li, X., Miguez, R., Tank, E. M. H., & Barmada, S. J.
Affiliations: University of Michigan (US)
Corresponding author: Barmada, Sami J.
ORCID: Bekier, Michael
Tank, Elizabeth M.H.
Barmada, Sami J.
Venue: Molecular Cell
Publisher: Elsevier BV
Published: 2023-01
Volume: 83
Issue: 2
Pages: 219-236.e7
ISSN: 1097-2765 (ISSN-L)
DOI: 10.1016/j.molcel.2022.12.019
PMC: PMC9899051
OpenAlex: W4315928462
Cited by: 112
References: 108
Citations by year: 2026: 31 · 2025: 34 · 2024: 32 · 2023: 15
FWCI: 18.26
Type: journal article
Language: en
Open access: bronze
doi.org
Licence: www.elsevier.com/tdm/userlicense/1.0/ (tdm)
www.elsevier.com/legal/tdmrep-license (tdm)
www.elsevier.com/open-access/userlicense/1.0/ (vor, 383 d)
doi.org/10.15223/policy-017 (stm-asf)
doi.org/10.15223/policy-037 (stm-asf)
doi.org/10.15223/policy-012 (stm-asf)
doi.org/10.15223/policy-029 (stm-asf)
doi.org/10.15223/policy-004 (stm-asf)
Locations: Molecular Cell (publishedVersion)
bioRxiv (Cold Spring Harbor Laboratory) (acceptedVersion)
PubMed (publishedVersion)
PubMed Central (acceptedVersion)
Funders: University of Michigan (P30AG072931, UL1TR000433)
A. Alfred Taubman Medical Research Institute
National Institutes of Health (1r56ns128110-01, UL1TR000433, CA04659229, P30CA04659229)
Michigan Institute for Clinical and Health Research (MICHR, UL1TR000433, UL1TR000433)
Rogel Cancer Center, University of Michigan
National Institute on Aging (R21AG075368, P30AG072931)
National Cancer Institute (P30CA046592)
National Institute of General Medical Sciences (T32GM007863, T32GM145470)
National Institute of Neurological Disorders and Stroke (R56 NS128110, R01NS113943, R01NS097542)
National Center for Advancing Translational Sciences
Robert Packard Center for ALS Research, Johns Hopkins University
Topics: Health Sciences › Medicine › Clinical Neurology › Amyotrophic Lateral Sclerosis Research
Life Sciences › Biochemistry, Genetics and Molecular Biology › Molecular Biology › RNA modifications and cancer
Life Sciences › Neuroscience › Cellular and Molecular Neuroscience › Alzheimer's disease research and treatments
Keywords: RNA methylation, N6-methyladenosine, TDP-43, amyotrophic lateral sclerosis, frontotemporal dementia, YTHDF2, RNA-binding proteins, RNA destabilization, neurodegeneration, spinal cord neurons
MeSH terms: Amyotrophic Lateral Sclerosis, DNA-Binding Proteins, Humans, Methylation, Neurons, RNA, Frontotemporal Dementia

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RNA methylation influences TDP43 binding and disease pathogenesis in models of amyotrophic lateral sclerosis and frontotemporal dementia

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