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Gmppb-mutant mice exhibit dystroglycanopathy symptoms that are rescued with GSK3β inhibition or AAV-mediated GMPPB gene replacement.

Overview

Authors: Ziwei Fu1, Tongchao Wang2, Chenyang Zhang1,3, Tianyu Qi1, Yanyan Chen1, Ju Yang4, Hua Yang1, Bing Yan5, Baoming Gong5, Weiqiao Lu6, Sushan Luo7, Ying Liu1, Lei Sun5,8, Hao Jiang6, Bo Chen9, Zhao Zhang3, Xiuping Liu4, Yuxiang Wang10
  1. Department of Pathology, School of Basic Medical Sciences, Fudan University,Shanghai, China
  2. Center for Food and Drug Inspection of NMPA,Beijing, 100163 China
  3. MOE Key Laboratory of Metabolism and Molecular Medicine, Department of Biochemistry and Molecular Biology, School of Basic Medical Sciences, Fudan University,Shanghai, China
  4. Department of Pathology, School of Basic Medical Sciences, Shanghai Fifth People’s Hospital, Fudan University,Shanghai, China
  5. Institute of Developmental Biology & Molecular Medicine, Fudan University,Shanghai, China
  6. Key Laboratory of Marine Drugs of Ministry of Education, Shandong Provincial Key Laboratory of Glycoscience and Glycotechnology, School of Medicine and Pharmacy, Ocean University of China,Qingdao, China
  7. Huashan Rare Disease Center and Department of Neurology, Huashan Hospital, Shanghai Medical College, National Center for Neurological Disorders, Fudan University,Shanghai, China
  8. State Key Laboratory of Genetic Engineering and National Center for International Research of Development and Disease, Institute of Developmental Biology and Molecular Medicine, Collaborative,Shanghai, China
  9. Keymed Biosciences Inc,Chengdu, China
  10. Department of Pathology, School of Basic Medical Sciences, Fudan University & Shanghai Pudong Hospital, Fudan University Pudong Medical Center,Shanghai, China
Journal: Nature communications, volume 17, issue 1, article 5340
Dates: received 4 August 2024; accepted 19 March 2026; published online 9 April 2026
Type: Research article · Language: English
License: CC BY-NC-ND
Identifiers: DOI 10.1038/s41467-026-71524-7 · PMID 41957353 · PMCID PMC13272650 · OpenAlex W7152406120
Open access: gold, a free copy (OpenAlex)
Status: data only
Categories: mouse (organism), other condition (population), cellular / molecular (subfield)
Methods: Preprocessing, Statistics, Smoothing, state filtering, decompositions, Evoked potentials, fMRI & imaging, Physiology & signal measures
Keywords: Neuromuscular disease, Disease model, Development, Mutation
MeSH: Dystroglycans*, Glycogen Synthase Kinase 3 beta*, Muscular Dystrophies*, Nucleotidyltransferases*, Animals, Disease Models, Animal, Female, Glycosylation, Male, Mice, Mice, Knockout, Muscle, Skeletal, Mutation, Wnt Signaling Pathway (* major topic)
Topic: Muscle Physiology and Disorders (Molecular Biology, Biochemistry, Genetics and Molecular Biology), according to OpenAlex
Funding: Shanghai Science and Technology Committee (20PJ1400700); Fudan University, JIF101074; Fundamental Research Funds for the Central Universities, 202261051 and 202262014
Citations: not cited yet (Europe PMC); 101 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

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Tracing map

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Data

Datasets cited

Data availability statement

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Read it in the paper: doi.org/10.1038/s41467-026-71524-7.

Versions

The history of this record: each version stored by the harvester or made by a correction of its authors or of the maintainers of its code, and what changed in its facts. The texts of the paper (its abstract, its availability statements) are not part of it; versions that changed only those are not listed.

Version 1, 29 September 2026: the first record

Recorded: type, language, journal, volume, issue, pages, dates, 18 authors, 4 keywords, 14 MeSH terms, 3 funders, 98 references.

Cite

This paper

Fu, Z., Wang, T., Zhang, C., Qi, T., Chen, Y., Yang, J., Yang, H., Yan, B., Gong, B., Lu, W., Luo, S., Liu, Y., Sun, L., Jiang, H., Chen, B., Zhang, Z., Liu, X., & Wang, Y. (2026). Gmppb-mutant mice exhibit dystroglycanopathy symptoms that are rescued with GSK3β inhibition or AAV-mediated GMPPB gene replacement. Nature communications, 17(1), 5340. https://doi.org/10.1038/s41467-026-71524-7

BibTeX

@article{fu2026gmppb,
author = {Fu, Ziwei and Wang, Tongchao and Zhang, Chenyang and Qi, Tianyu and Chen, Yanyan and Yang, Ju and Yang, Hua and Yan, Bing and Gong, Baoming and Lu, Weiqiao and Luo, Sushan and Liu, Ying and Sun, Lei and Jiang, Hao and Chen, Bo and Zhang, Zhao and Liu, Xiuping and Wang, Yuxiang},
title = {{Gmppb-mutant mice exhibit dystroglycanopathy symptoms that are rescued with GSK3β inhibition or AAV-mediated GMPPB gene replacement}},
journal = {Nature communications},
year = {2026},
month = apr,
volume = {17},
number = {1},
pages = {5340},
publisher = {Nature Publishing Group},
issn = {2041-1723},
doi = {10.1038/s41467-026-71524-7},
url = {https://doi.org/10.1038/s41467-026-71524-7},
pmid = {41957353},
pmcid = {PMC13272650}
}

RIS

TY - JOUR
AU - Fu, Ziwei
AU - Wang, Tongchao
AU - Zhang, Chenyang
AU - Qi, Tianyu
AU - Chen, Yanyan
AU - Yang, Ju
AU - Yang, Hua
AU - Yan, Bing
AU - Gong, Baoming
AU - Lu, Weiqiao
AU - Luo, Sushan
AU - Liu, Ying
AU - Sun, Lei
AU - Jiang, Hao
AU - Chen, Bo
AU - Zhang, Zhao
AU - Liu, Xiuping
AU - Wang, Yuxiang
TI - Gmppb-mutant mice exhibit dystroglycanopathy symptoms that are rescued with GSK3β inhibition or AAV-mediated GMPPB gene replacement
T2 - Nature communications
J2 - Nat Commun
PY - 2026
DA - 2026/04/09
VL - 17
IS - 1
SP - 5340
SN - 2041-1723
PB - Nature Publishing Group
DO - 10.1038/s41467-026-71524-7
UR - https://doi.org/10.1038/s41467-026-71524-7
LA - en
ER -

CSL-JSON

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"language": "en",
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