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Restoring early postnatal synaptic dysregulation rescues motor neuron degeneration in a mouse model of Spinal and Bulbar Muscular Atrophy.

Overview

Authors: Tomoki Hirunagi1,2, Kentaro Sahashi1, Madoka Iida1, Kazunari Onodera1,3, Satoshi Yokoi1,4, Yosuke Ogura1, Genki Tohnai1,5, Kenji Sakakibara1, Kentaro Maeda1, C Frank Bennett6, Yohei Okada3, Masahisa Katsuno1
ORCID iDs: Tomoki Hirunagi, Kentaro Sahashi, Madoka Iida, Yosuke Ogura, Genki Tohnai, Yohei Okada, Masahisa Katsuno
  1. Department of Neurology, Nagoya University Graduate School of Medicine, Nagoya, Japan
  2. Nagoya University Institute for Advanced Research, Nagoya, Japan
  3. Department of Neural iPSC Research, Institute for Medical Science of Aging, Aichi Medical University School of Medicine, Nagakute, Japan
  4. Department of Pathophysiological Laboratory Sciences, Nagoya University Graduate School of Medicine, Nagoya, Japan
  5. Division of ALS Research, Aichi Medical University, Nagakute, Japan
  6. Ionis Pharmaceuticals, Carlsbad, CA USA
Institutions: Nagoya University (Japan); Aichi Medical University (Japan); Ionis Pharmaceuticals (United States) (United States)
Journal: Nature communications, volume 17, issue 1, article 2412
Dates: received 4 June 2024; accepted 23 February 2026; published online 27 March 2026
Type: Research article · Language: English
License: CC BY-NC-ND
Identifiers: DOI 10.1038/s41467-026-70244-2 · PMID 41896536 · PMCID PMC13031292 · OpenAlex W7141147261
Open access: gold, a free copy (OpenAlex)
Status: empty repository (how the code is found and verified)
Categories: human (organism), mouse (organism), other condition (population), cellular / molecular (subfield)
Methods: Statistics, Smoothing, state filtering, decompositions, Connectivity, Single-unit activity, calcium imaging
Keywords: Neurodegeneration, Neuromuscular disease, Motor neuron disease
MeSH: Motor Neurons*, Muscular Atrophy, Spinal*, Nerve Degeneration*, Receptors, Androgen*, Synapses*, Animals, Disease Models, Animal, Female, Humans, Induced Pluripotent Stem Cells, Male, Mice, Mice, Transgenic, Oligonucleotides, Antisense, Peptides, Repressor Proteins (* major topic)
Topic: Genetic Neurodegenerative Diseases (Cellular and Molecular Neuroscience, Neuroscience), according to OpenAlex
Funding: Japan Agency for Medical Research and Development (AMED) (JP22bm0804020, JP22am0401007, JP22nk0101575, P25bm1423003); MEXT | Japan Society for the Promotion of Science (JSPS) (JP24K18683, JP24K18712, JP25K02585, JP23K24249, JP20H00527, JP23H00420)
Citations: not cited yet (Europe PMC); 79 references in the paper

Abstract

The abstract is not reproduced here: the paper's license (CC BY-NC-ND) does not allow it. Read it in the paper, at the publisher or on Europe PMC.

Code

No file of the authors' code could be read here: it is described below, and read at its source.

waterriding-design/hirunagi-et-al.-2025

License: none: the authors keep all their rights
State: the link answers, verified on 30 September 2026
Evidence: files inventoried
Commit: 5d571b3f1d92c786eda22d630ae21cf8e86143f6, 29 November 2025
Size: 1 file, 0 scripts
Software Heritage: not archived
Found in: “Code availability”
Not found: README, license file, CITATION.cff, environment file, tests, continuous integration, documentation
Availability: 1 check, the latest on 30 September 2026: the link answers
  • 30 September 2026: the link answers
At the source: github.com/waterriding-design/Hirunagi-et-al.-2025/blob/main/snRNA-seq2025

Code availability statement

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Read it in the paper: doi.org/10.1038/s41467-026-70244-2.

Tracing map

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Data

Datasets cited

Data availability statement

The paper has a data availability statement. Its license (CC BY-NC-ND) does not allow reproducing it here; in short, from what the harvester recognized in it:

Read it in the paper: doi.org/10.1038/s41467-026-70244-2.

Versions

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Version 1, 30 September 2026: the first record

Recorded: type, language, journal, volume, issue, pages, dates, 12 authors, 3 keywords, 16 MeSH terms, 2 funders, 79 references.

Cite

This paper

Hirunagi, T., Sahashi, K., Iida, M., Onodera, K., Yokoi, S., Ogura, Y., Tohnai, G., Sakakibara, K., Maeda, K., Bennett, C. F., Okada, Y., & Katsuno, M. (2026). Restoring early postnatal synaptic dysregulation rescues motor neuron degeneration in a mouse model of Spinal and Bulbar Muscular Atrophy. Nature communications, 17(1), 2412. https://doi.org/10.1038/s41467-026-70244-2

BibTeX

@article{hirunagi2026restoring,
author = {Hirunagi, Tomoki and Sahashi, Kentaro and Iida, Madoka and Onodera, Kazunari and Yokoi, Satoshi and Ogura, Yosuke and Tohnai, Genki and Sakakibara, Kenji and Maeda, Kentaro and Bennett, C Frank and Okada, Yohei and Katsuno, Masahisa},
title = {{Restoring early postnatal synaptic dysregulation rescues motor neuron degeneration in a mouse model of Spinal and Bulbar Muscular Atrophy}},
journal = {Nature communications},
year = {2026},
month = mar,
volume = {17},
number = {1},
pages = {2412},
publisher = {Nature Publishing Group},
issn = {2041-1723},
doi = {10.1038/s41467-026-70244-2},
url = {https://doi.org/10.1038/s41467-026-70244-2},
pmid = {41896536},
pmcid = {PMC13031292}
}

RIS

TY - JOUR
AU - Hirunagi, Tomoki
AU - Sahashi, Kentaro
AU - Iida, Madoka
AU - Onodera, Kazunari
AU - Yokoi, Satoshi
AU - Ogura, Yosuke
AU - Tohnai, Genki
AU - Sakakibara, Kenji
AU - Maeda, Kentaro
AU - Bennett, C Frank
AU - Okada, Yohei
AU - Katsuno, Masahisa
TI - Restoring early postnatal synaptic dysregulation rescues motor neuron degeneration in a mouse model of Spinal and Bulbar Muscular Atrophy
T2 - Nature communications
J2 - Nat Commun
PY - 2026
DA - 2026/03/27
VL - 17
IS - 1
SP - 2412
SN - 2041-1723
PB - Nature Publishing Group
DO - 10.1038/s41467-026-70244-2
UR - https://doi.org/10.1038/s41467-026-70244-2
LA - en
ER -

CSL-JSON

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