Nature Communications (Oct 2022)
Rescue of deficits by Brwd1 copy number restoration in the Ts65Dn mouse model of Down syndrome
- Sasha L. Fulton,
- Wendy Wenderski,
- Ashley E. Lepack,
- Andrew L. Eagle,
- Tomas Fanutza,
- Ryan M. Bastle,
- Aarthi Ramakrishnan,
- Emma C. Hays,
- Arianna Neal,
- Jaroslav Bendl,
- Lorna A. Farrelly,
- Amni Al-Kachak,
- Yang Lyu,
- Bulent Cetin,
- Jennifer C. Chan,
- Tina N. Tran,
- Rachael L. Neve,
- Randall J. Roper,
- Kristen J. Brennand,
- Panos Roussos,
- John C. Schimenti,
- Allyson K. Friedman,
- Li Shen,
- Robert D. Blitzer,
- Alfred J. Robison,
- Gerald R. Crabtree,
- Ian Maze
Affiliations
- Sasha L. Fulton
- Nash Family Department of Neuroscience, Friedman Brain Institute, Icahn School of Medicine at Mount Sinai
- Wendy Wenderski
- Department of Pathology, Stanford Medical School
- Ashley E. Lepack
- Nash Family Department of Neuroscience, Friedman Brain Institute, Icahn School of Medicine at Mount Sinai
- Andrew L. Eagle
- Department of Physiology, Michigan State University
- Tomas Fanutza
- Department of Pharmacological Sciences, Icahn School of Medicine at Mount Sinai
- Ryan M. Bastle
- Nash Family Department of Neuroscience, Friedman Brain Institute, Icahn School of Medicine at Mount Sinai
- Aarthi Ramakrishnan
- Nash Family Department of Neuroscience, Friedman Brain Institute, Icahn School of Medicine at Mount Sinai
- Emma C. Hays
- Nash Family Department of Neuroscience, Friedman Brain Institute, Icahn School of Medicine at Mount Sinai
- Arianna Neal
- Nash Family Department of Neuroscience, Friedman Brain Institute, Icahn School of Medicine at Mount Sinai
- Jaroslav Bendl
- Department of Psychiatry, Icahn School of Medicine at Mount Sinai
- Lorna A. Farrelly
- Nash Family Department of Neuroscience, Friedman Brain Institute, Icahn School of Medicine at Mount Sinai
- Amni Al-Kachak
- Nash Family Department of Neuroscience, Friedman Brain Institute, Icahn School of Medicine at Mount Sinai
- Yang Lyu
- Nash Family Department of Neuroscience, Friedman Brain Institute, Icahn School of Medicine at Mount Sinai
- Bulent Cetin
- Nash Family Department of Neuroscience, Friedman Brain Institute, Icahn School of Medicine at Mount Sinai
- Jennifer C. Chan
- Nash Family Department of Neuroscience, Friedman Brain Institute, Icahn School of Medicine at Mount Sinai
- Tina N. Tran
- Department of Biomedical Sciences, Cornell University
- Rachael L. Neve
- McGovern Institute for Brain Research, Massachusetts Institute of Technology
- Randall J. Roper
- Department of Biology, Indiana University-Purdue University
- Kristen J. Brennand
- Nash Family Department of Neuroscience, Friedman Brain Institute, Icahn School of Medicine at Mount Sinai
- Panos Roussos
- Department of Psychiatry, Icahn School of Medicine at Mount Sinai
- John C. Schimenti
- Department of Biomedical Sciences, Cornell University
- Allyson K. Friedman
- Department of Biological Sciences, City University of New York-Hunter College
- Li Shen
- Nash Family Department of Neuroscience, Friedman Brain Institute, Icahn School of Medicine at Mount Sinai
- Robert D. Blitzer
- Department of Pharmacological Sciences, Icahn School of Medicine at Mount Sinai
- Alfred J. Robison
- Department of Physiology, Michigan State University
- Gerald R. Crabtree
- Department of Pathology, Stanford Medical School
- Ian Maze
- Nash Family Department of Neuroscience, Friedman Brain Institute, Icahn School of Medicine at Mount Sinai
- DOI
- https://doi.org/10.1038/s41467-022-34200-0
- Journal volume & issue
-
Vol. 13,
no. 1
pp. 1 – 17
Abstract
The molecular mechanisms underlying deficits in Down syndrome remain unclear. Here, the authors show that copy number restoration of a chromatin remodeler in trisomic mice is sufficient to rescue epigenomic, physiological and cognitive deficits.
