ArticleiScience2021
An integrated multi-omic analysis of iPSC-derived motor neurons from C9ORF72 ALS patients.
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Article in iScience, 2021. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 37 papers.
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37citing papers in PubMed
3.7field-weighted citation impact, top 5% of its field
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Who cites it
37 citing papers in PubMed, 48 citations in OpenAlex.
- Neurotropic alphavirus infection induces PARP-1 hyperactivation-mediated energy collapse in motor neurons.Journal of virology · 2026Article
- Article
- Pharmacological rescue of mitochondrial dysfunction, neurite degeneration, and premature death of ALS and AD iPSC-derived neurons.bioRxiv : the preprint server for biology · 2026Article
- Repeat expansions inbioRxiv : the preprint server for biology · 2026Article
- Review
- Smarter stem cells: how AI is supercharging iPSC technology.Cell and tissue research · 2025Review
- An alternative cytoplasmic SFPQ isoform with reduced phase separation potential is up-regulated in ALS.Science advances · 2025Article
- Ropinirole Functions Through a Dopamine Receptor D2-Independent Mechanism to Ameliorate Amyotrophic Lateral Sclerosis Phenotypes in TARDBP-Mutant iPSC-Derived Motor Neurons.Journal of neurochemistry · 2025Article
- An organ-chip model of sporadic ALS using iPSC-derived spinal cord motor neurons and an integrated blood-brain-like barrier.Cell stem cell · 2025Article
- Intersecting impact of CAG repeat and huntingtin knockout in stem cell-derived cortical neurons.Neurobiology of disease · 2025Article
- An integrative systems-biology approach defines mechanisms of Alzheimer's disease neurodegeneration.Nature communications · 2025Article
- CellPHIE: Integrating Pathway Discovery With Pooled Profiling of Perturbations Uncovers Pathways of Huntington's Disease, Including Genetic Modifiers of Neuronal Development and Morphology.bioRxiv : the preprint server for biology · 2025Article
- Review
- Amyotrophic lateral sclerosis caused by hexanucleotide repeat expansions in C9orf72: from genetics to therapeutics.The Lancet. Neurology · 2025Review
- Opposing roles of p38α-mediated phosphorylation and PRMT1-mediated arginine methylation in driving TDP-43 proteinopathy.Cell reports · 2025Article
- Nuclear pore dysfunction and disease: a complex opportunity.Nucleus (Austin, Tex.) · 2024Review
- Expanded ATXN1 alters transcription and calcium signaling in SCA1 human motor neurons differentiated from induced pluripotent stem cells.Neurobiology of disease · 2024Article
- Poly-GR repeats associated with ALS/FTD geneScience signaling · 2024Article
- Multiomics Evaluation of Human iPSCs and iPSC-Derived Neurons.Journal of proteome research · 2024Article
- Identifying dysregulated regions in amyotrophic lateral sclerosis through chromatin accessibility outliers.HGG advances · 2024Article
Corrections and comments
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Authors and funding
48 authors at 5 institutions in 1 country.
NeuroLINCS Consortium
Jonathan LiDepartment of Biological Engineering, Massachusetts Institute of Technology, Cambridge, MA 02139, USA.
Ryan G LimUCI MIND, University of California, Irvine, CA 92697, USA.
Julia A KayeCenter for Systems and Therapeutics and the Taube/Koret Center for Neurodegenerative Disease, Gladstone Institutes, University of California, San Francisco, San Francisco, CA 94158, USA.
Victoria DardovThe Board of Governors Regenerative Medicine Institute, Cedars-Sinai Medical Center, 8700 Beverly Boulevard, Los Angeles, CA 90048, USA.
Alyssa N CoyneBrain Science Institute, Johns Hopkins University School of Medicine, Baltimore, MA 212056, USA.
Jie WuDepartment of Biological Chemistry, University of California, Irvine, CA 92697, USA.
Pamela MilaniDepartment of Biological Engineering, Massachusetts Institute of Technology, Cambridge, MA 02139, USA.
Andrew ChengCellular and Molecular Medicine Program, Johns Hopkins University School of Medicine, Baltimore, MA 212056, USA.
Terri G ThompsonUCI MIND, University of California, Irvine, CA 92697, USA.
Loren OrnelasThe Board of Governors Regenerative Medicine Institute, Cedars-Sinai Medical Center, 8700 Beverly Boulevard, Los Angeles, CA 90048, USA.
Aaron FrankThe Board of Governors Regenerative Medicine Institute, Cedars-Sinai Medical Center, 8700 Beverly Boulevard, Los Angeles, CA 90048, USA.
Miriam AdamDepartment of Biological Engineering, Massachusetts Institute of Technology, Cambridge, MA 02139, USA.
Maria G BanuelosThe Board of Governors Regenerative Medicine Institute, Cedars-Sinai Medical Center, 8700 Beverly Boulevard, Los Angeles, CA 90048, USA.
Malcolm CasaleUCI MIND, University of California, Irvine, CA 92697, USA.
Veerle CoxCellular and Molecular Medicine Program, Johns Hopkins University School of Medicine, Baltimore, MA 212056, USA.
Renan Escalante-ChongDepartment of Biological Engineering, Massachusetts Institute of Technology, Cambridge, MA 02139, USA.
J Gavin DaigleBrain Science Institute, Johns Hopkins University School of Medicine, Baltimore, MA 212056, USA.
Emilda GomezThe Board of Governors Regenerative Medicine Institute, Cedars-Sinai Medical Center, 8700 Beverly Boulevard, Los Angeles, CA 90048, USA.
Lindsey HayesDepartment of Neurology and Neuroscience, Johns Hopkins University School of Medicine, Baltimore, MA 212056, USA.
Ronald HolewenskiAdvanced Clinical Biosystems Research Institute, The Barbra Streisand Heart Center, The Smidt Heart Institute, Cedars-Sinai Medical Center, Los Angeles, CA, USA.
Susan LeiThe Board of Governors Regenerative Medicine Institute, Cedars-Sinai Medical Center, 8700 Beverly Boulevard, Los Angeles, CA 90048, USA.
Alex LenailDepartment of Biological Engineering, Massachusetts Institute of Technology, Cambridge, MA 02139, USA.
Leandro LimaCenter for Systems and Therapeutics and the Taube/Koret Center for Neurodegenerative Disease, Gladstone Institutes, University of California, San Francisco, San Francisco, CA 94158, USA.
Berhan MandefroThe Board of Governors Regenerative Medicine Institute, Cedars-Sinai Medical Center, 8700 Beverly Boulevard, Los Angeles, CA 90048, USA.
Andrea MatlockAdvanced Clinical Biosystems Research Institute, The Barbra Streisand Heart Center, The Smidt Heart Institute, Cedars-Sinai Medical Center, Los Angeles, CA, USA.
Lindsay PantherThe Board of Governors Regenerative Medicine Institute, Cedars-Sinai Medical Center, 8700 Beverly Boulevard, Los Angeles, CA 90048, USA.
Natasha Leanna Patel-MurrayDepartment of Biological Engineering, Massachusetts Institute of Technology, Cambridge, MA 02139, USA.
Jacqueline PhamDepartment of Neurology and Neuroscience, Johns Hopkins University School of Medicine, Baltimore, MA 212056, USA.
Divya RamamoorthyDepartment of Biological Engineering, Massachusetts Institute of Technology, Cambridge, MA 02139, USA.
Karen SachsDepartment of Biological Engineering, Massachusetts Institute of Technology, Cambridge, MA 02139, USA.
Brandon ShelleyThe Board of Governors Regenerative Medicine Institute, Cedars-Sinai Medical Center, 8700 Beverly Boulevard, Los Angeles, CA 90048, USA.
Jennifer StocksdaleUCI MIND, University of California, Irvine, CA 92697, USA.
Hannah TrostThe Board of Governors Regenerative Medicine Institute, Cedars-Sinai Medical Center, 8700 Beverly Boulevard, Los Angeles, CA 90048, USA.
Mark WilhelmBrain Science Institute, Johns Hopkins University School of Medicine, Baltimore, MA 212056, USA.
Vidya VenkatramanAdvanced Clinical Biosystems Research Institute, The Barbra Streisand Heart Center, The Smidt Heart Institute, Cedars-Sinai Medical Center, Los Angeles, CA, USA.
Brook T WassieDepartment of Biological Engineering, Massachusetts Institute of Technology, Cambridge, MA 02139, USA.
Stacia WymanSue and Bill Gross Stem Cell Center, University of California, Irvine, CA 92697, USA.
Stephanie YangBrain Science Institute, Johns Hopkins University School of Medicine, Baltimore, MA 212056, USA.
NYGC ALS Consortium
Jennifer E Van EykAdvanced Clinical Biosystems Research Institute, The Barbra Streisand Heart Center, The Smidt Heart Institute, Cedars-Sinai Medical Center, Los Angeles, CA, USA.
Thomas E LloydDepartment of Neurology and Neuroscience, Johns Hopkins University School of Medicine, Baltimore, MA 212056, USA.
Steven FinkbeinerCenter for Systems and Therapeutics and the Taube/Koret Center for Neurodegenerative Disease, Gladstone Institutes, University of California, San Francisco, San Francisco, CA 94158, USA.
Ernest FraenkelDepartment of Biological Engineering, Massachusetts Institute of Technology, Cambridge, MA 02139, USA.
Jeffrey D RothsteinBrain Science Institute, Johns Hopkins University School of Medicine, Baltimore, MA 212056, USA.
Dhruv SareenThe Board of Governors Regenerative Medicine Institute, Cedars-Sinai Medical Center, 8700 Beverly Boulevard, Los Angeles, CA 90048, USA.
Clive N SvendsenThe Board of Governors Regenerative Medicine Institute, Cedars-Sinai Medical Center, 8700 Beverly Boulevard, Los Angeles, CA 90048, USA.
Leslie M ThompsonUCI MIND, University of California, Irvine, CA 92697, USA.
Cedars-Sinai Medical Center · USMassachusetts Institute of Technology · USJohns Hopkins University · USUniversity of California, Irvine · USGladstone Institutes · US
Funding
Univ.of Calif., Irvine Cancer Center Support GrantP30CA062203 · NCI · UNIVERSITY OF CALIFORNIA-IRVINE · PI Melanie Funes · 1994 to 2026
$57.9MNeuron and Glial Cellular Signatures from Normal and Diseased iPS Cells-SupplementU54NS091046 · NINDS · UNIVERSITY OF CALIFORNIA-IRVINE · PI FINKBEINER, STEVEN M, FRAENKEL, ERNEST · 2014 to 2020
$11.7MEpigenetic Pathology and Therapy in Huntington's DiseaseR01NS089076 · NINDS · MASSACHUSETTS INSTITUTE OF TECHNOLOGY · PI FRAENKEL, ERNEST, HOUSMAN, DAVID · 2015 to 2024
$4.9MGraduate Training in Computational and Systems BiologyT32GM087237 · NIGMS · MASSACHUSETTS INSTITUTE OF TECHNOLOGY · PI BURGE, CHRISTOPHER B · 2009 to 2023
$4.6MNucleocytoplasmic transport and nuclear pore disruption in ALS/FTDR01NS094239 · NINDS · JOHNS HOPKINS UNIVERSITY · PI LLOYD, THOMAS E., ROTHSTEIN, JEFFREY D · 2016 to 2020
$3.4MALS/FTD mutant C9orf72-induced genetic and nuclear pathology in iPS cell modelsR01NS085207 · NINDS · JOHNS HOPKINS UNIVERSITY · PI ROTHSTEIN, JEFFREY D, SATTLER, RITA · 2013 to 2017
$1.8MFG-nucleoporins and nuclear transport disruption in C9ORF72-ALS/FTDK08NS104273 · NINDS · JOHNS HOPKINS UNIVERSITY · PI HAYES, LINDSEY RENAE · 2017 to 2021
$999kNCI NIH HHS P30 CA062203NIGMS NIH HHS T32 GM087237NINDS NIH HHS K08 NS104273NINDS NIH HHS R01 NS085207NINDS NIH HHS R01 NS089076NINDS NIH HHS R01 NS094239NINDS NIH HHS U54 NS091046
Abstract
Neurodegenerative diseases are challenging for systems biology because of the lack of reliable animal models or patient samples at early disease stages. Induced pluripotent stem cells (iPSCs) could address these challenges. We investigated DNA, RNA, epigenetics, and proteins in iPSC-derived motor neurons from patients with ALS carrying hexanucleotide expansions in
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Biological sciencesNeuroscienceOmicsSystems biologySystems neuroscience
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