The Multiple Functions of Fibrillin-1 Microfibrils in Organismal Physiology.
Marfan syndrome
bone lengthening
dilated cardiomyopathy
fibrillin-1
lens dislocation
osteopenia
thoracic aortic aneurysm
Journal
International journal of molecular sciences
ISSN: 1422-0067
Titre abrégé: Int J Mol Sci
Pays: Switzerland
ID NLM: 101092791
Informations de publication
Date de publication:
08 Feb 2022
08 Feb 2022
Historique:
received:
17
12
2021
revised:
27
01
2022
accepted:
03
02
2022
entrez:
15
2
2022
pubmed:
16
2
2022
medline:
5
3
2022
Statut:
epublish
Résumé
Fibrillin-1 is the major structural component of the 10 nm-diameter microfibrils that confer key physical and mechanical properties to virtually every tissue, alone and together with elastin in the elastic fibers. Mutations in fibrillin-1 cause pleiotropic manifestations in Marfan syndrome (MFS), including dissecting thoracic aortic aneurysms, myocardial dysfunction, progressive bone loss, disproportionate skeletal growth, and the dislocation of the crystalline lens. The characterization of these MFS manifestations in mice, that replicate the human phenotype, have revealed that the underlying mechanisms are distinct and organ-specific. This brief review summarizes relevant findings supporting this conclusion.
Identifiants
pubmed: 35163812
pii: ijms23031892
doi: 10.3390/ijms23031892
pmc: PMC8836826
pii:
doi:
Substances chimiques
FBN1 protein, human
0
Fbn1 protein, mouse
0
Fibrillin-1
0
Types de publication
Journal Article
Review
Langues
eng
Sous-ensembles de citation
IM
Subventions
Organisme : NIH HHS
ID : HL126173
Pays : United States
Organisme : Marfan Foundation
ID : N/A
Organisme : NIAMS NIH HHS
ID : R01 AR069307
Pays : United States
Organisme : NIH HHS
ID : HL134605
Pays : United States
Organisme : NIH HHS
ID : AR069307
Pays : United States
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