Mapping brain networks in MPS I mice and their restoration following gene therapy.
Journal
Scientific reports
ISSN: 2045-2322
Titre abrégé: Sci Rep
Pays: England
ID NLM: 101563288
Informations de publication
Date de publication:
05 08 2023
05 08 2023
Historique:
received:
09
02
2023
accepted:
02
08
2023
medline:
7
8
2023
pubmed:
6
8
2023
entrez:
5
8
2023
Statut:
epublish
Résumé
Mucopolysaccharidosis type I (MPS I) is an inherited lysosomal disorder that causes syndromes characterized by physiological dysfunction in many organs and tissues. Despite the recognizable morphological and behavioral deficits associated with MPS I, neither the underlying alterations in functional neural connectivity nor its restoration following gene therapy have been shown. By employing high-resolution resting-state fMRI (rs-fMRI), we found significant reductions in functional neural connectivity in the limbic areas of the brain that play key roles in learning and memory in MPS I mice, and that adeno-associated virus (AAV)-mediated gene therapy can reestablish most brain connectivity. Using logistic regression in MPS I and treated animals, we identified functional networks with the most alterations. The rs-fMRI and statistical methods should be translatable into clinical evaluation of humans with neurological disorders.
Identifiants
pubmed: 37543633
doi: 10.1038/s41598-023-39939-0
pii: 10.1038/s41598-023-39939-0
pmc: PMC10404260
doi:
Types de publication
Journal Article
Research Support, Non-U.S. Gov't
Research Support, N.I.H., Extramural
Langues
eng
Sous-ensembles de citation
IM
Pagination
12716Subventions
Organisme : NIBIB NIH HHS
ID : U01 EB026978
Pays : United States
Organisme : NIH HHS
ID : R01 NS118330
Pays : United States
Organisme : NINDS NIH HHS
ID : R01 NS133006
Pays : United States
Organisme : NIMH NIH HHS
ID : R01 MH111413
Pays : United States
Organisme : NIBIB NIH HHS
ID : P41 EB027061
Pays : United States
Organisme : NINDS NIH HHS
ID : R01 NS118330
Pays : United States
Informations de copyright
© 2023. Springer Nature Limited.
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