PDGFRα signaling regulates cartilage and fibrous tissue differentiation during synovial joint development

Abstract Synovial joints develop from mesenchymal structures called interzones, with progenitor cells differentiating into specialized cartilaginous and fibrous tissues of the joint. Platelet-derived growth factor receptor-α (PDGFRα) is a tyrosine kinase expressed by cells of the limb bud, but its r...

Full description

Saved in:
Bibliographic Details
Main Authors: John P. Woods, Alex Rackley, Hae Ryong Kwon, Lorin E. Olson
Format: Article
Language:English
Published: Nature Portfolio 2025-04-01
Series:Nature Communications
Online Access:https://doi.org/10.1038/s41467-025-59207-1
Tags: Add Tag
No Tags, Be the first to tag this record!
_version_ 1850206514617253888
author John P. Woods
Alex Rackley
Hae Ryong Kwon
Lorin E. Olson
author_facet John P. Woods
Alex Rackley
Hae Ryong Kwon
Lorin E. Olson
author_sort John P. Woods
collection DOAJ
description Abstract Synovial joints develop from mesenchymal structures called interzones, with progenitor cells differentiating into specialized cartilaginous and fibrous tissues of the joint. Platelet-derived growth factor receptor-α (PDGFRα) is a tyrosine kinase expressed by cells of the limb bud, but its role in limb development is unknown. To investigate PDGFRα function, we generated mice expressing mutant PDGFRα with a point mutation (D842V) that increases receptor signaling. Mutant hindlimbs are immobile with knee joints fused by cartilage and lacking ligaments and menisci. The interzone marker Gdf5 is initially expressed at E12.5 but is downregulated thereafter, suggesting a defect in interzone maintenance. Omics analysis of the joint tissues identifies ectopic cartilage matrix expressing genes for cartilage and fibrotic tissue. Thus, elevated PDGFRα signaling corrupts joint development by downregulating Gdf5 and redirecting interzone progenitors into a fibrocartilage fate. This suggests that tight regulation of tyrosine kinase activity is necessary for the development of the mouse knee joint.
format Article
id doaj-art-0482479c25ae4e0184c9e4b818b631da
institution OA Journals
issn 2041-1723
language English
publishDate 2025-04-01
publisher Nature Portfolio
record_format Article
series Nature Communications
spelling doaj-art-0482479c25ae4e0184c9e4b818b631da2025-08-20T02:10:49ZengNature PortfolioNature Communications2041-17232025-04-0116111710.1038/s41467-025-59207-1PDGFRα signaling regulates cartilage and fibrous tissue differentiation during synovial joint developmentJohn P. Woods0Alex Rackley1Hae Ryong Kwon2Lorin E. Olson3Cardiovascular Biology Research Program, Oklahoma Medical Research FoundationCardiovascular Biology Research Program, Oklahoma Medical Research FoundationCardiovascular Biology Research Program, Oklahoma Medical Research FoundationCardiovascular Biology Research Program, Oklahoma Medical Research FoundationAbstract Synovial joints develop from mesenchymal structures called interzones, with progenitor cells differentiating into specialized cartilaginous and fibrous tissues of the joint. Platelet-derived growth factor receptor-α (PDGFRα) is a tyrosine kinase expressed by cells of the limb bud, but its role in limb development is unknown. To investigate PDGFRα function, we generated mice expressing mutant PDGFRα with a point mutation (D842V) that increases receptor signaling. Mutant hindlimbs are immobile with knee joints fused by cartilage and lacking ligaments and menisci. The interzone marker Gdf5 is initially expressed at E12.5 but is downregulated thereafter, suggesting a defect in interzone maintenance. Omics analysis of the joint tissues identifies ectopic cartilage matrix expressing genes for cartilage and fibrotic tissue. Thus, elevated PDGFRα signaling corrupts joint development by downregulating Gdf5 and redirecting interzone progenitors into a fibrocartilage fate. This suggests that tight regulation of tyrosine kinase activity is necessary for the development of the mouse knee joint.https://doi.org/10.1038/s41467-025-59207-1
spellingShingle John P. Woods
Alex Rackley
Hae Ryong Kwon
Lorin E. Olson
PDGFRα signaling regulates cartilage and fibrous tissue differentiation during synovial joint development
Nature Communications
title PDGFRα signaling regulates cartilage and fibrous tissue differentiation during synovial joint development
title_full PDGFRα signaling regulates cartilage and fibrous tissue differentiation during synovial joint development
title_fullStr PDGFRα signaling regulates cartilage and fibrous tissue differentiation during synovial joint development
title_full_unstemmed PDGFRα signaling regulates cartilage and fibrous tissue differentiation during synovial joint development
title_short PDGFRα signaling regulates cartilage and fibrous tissue differentiation during synovial joint development
title_sort pdgfrα signaling regulates cartilage and fibrous tissue differentiation during synovial joint development
url https://doi.org/10.1038/s41467-025-59207-1
work_keys_str_mv AT johnpwoods pdgfrasignalingregulatescartilageandfibroustissuedifferentiationduringsynovialjointdevelopment
AT alexrackley pdgfrasignalingregulatescartilageandfibroustissuedifferentiationduringsynovialjointdevelopment
AT haeryongkwon pdgfrasignalingregulatescartilageandfibroustissuedifferentiationduringsynovialjointdevelopment
AT lorineolson pdgfrasignalingregulatescartilageandfibroustissuedifferentiationduringsynovialjointdevelopment