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Repairing Gli-tches during bone growth

Gli1+ fetal cells give rise to long-lived cartilage progenitors in the long bones. While normally dormant until postnatally, they expand upon (and compensate for) fetal growth perturbations, arising in part from external Pdgfra+ cells.

Long bones in the limbs grow via a cartilage template that is progressively replaced by bone. In mice, cartilage progenitors show a postnatal switch of behaviour, from being short-lived to being self-renewing, long-lived cartilage progenitors (LLCPs). A big question in the field is which cells in the fetal limb are precursors of LLCPs, and when/how LLCPs are generated. In this study, the HFSP Career Development Award recipient Alberto Rosello-Diez and his team start to provide an answer, showing that cells that express the transcription factor GLI1 are the fetal precursors of postnatal LLCPs, and that Gli1+ LLCP precursors remain mostly dormant until postnatal stages.

However, in response to genetic inhibition of cell proliferation in fetal cartilage, the descendants of Gli1+ cells dramatically increase their numbers in the cartilage, compensating for the perturbation and enabling normal growth. Specific elimination of the cartilage cells (but not other cell types) derived from Gli1+ precursors precludes the compensation for the genetic inhibition of cell proliferation.

Fetal tibia stained for Gli1 lineage in red, pulse-chased cartilage cells in green, nuclei in blue. Credit: Xinli Qu

 

The HFSP Awardee further shows that some reparative Gli1+ cells originate from Pdgfra+ cells located outside the cartilage, revealing the surrounding tissues as a source of back-up cartilage progenitors when needed. While the exact process of conversion and/or migration from non-cartilage Pdgfra+ cells (P) to Gli1+ cartilage cells (G) has not been completely elucidated, it seems to involve a self-regulating mechanism. If the cartilage is growing well, it produces a substance that blocks the P-to-G conversion. If growth falters, however, that substance is produced at lower levels, lifting the brakes on the P-to-G conversion.

While several new questions have been raised by this study, it sheds light on the long-standing question of the origin and regulation of LLCPs, laying the groundwork for future approaches to stimulate the in vivo production of cartilage-reparative cells to treat congenital or acquired growth disorders.

Reference

Gli1-expressing stromal cells are highly reparative precursors of long-lived chondroprogenitors in the fetal murine limb. Xinli Qu, Ehsan Razmara, Ashiq Khader C, Chee Ho H’ng, Kailash K. Vinu, Luciano G. Martelotto, Maia Zethoven, Fernando J. Rossello, Shanika L. Amarasinghe, David R. Powell & Alberto Rosello-Diez. Nature Communications volume 16, Article number: 10107 (2025). https://doi.org/10.1038/s41467-025-65029-y

Other references

HFSP Reference: LT000661/2020-L
HFSP Career Development Awardee: Alberto Rosello-Diez, Sloan Kettering Institute, USA; Host Supervisor Alexandra Joyner.