Molecular analysis of enthesopathy in a mouse model of hypophosphatemic rickets

Development
Eva S LiuMarie B Demay

Abstract

The bone tendon attachment site known as the enthesis comprises a transitional zone between bone and tendon, and plays an important role in enabling movement at this site. X-linked hypophosphatemia (XLH) is characterized by impaired activation of vitamin D, elevated serum FGF23 levels and low serum phosphate levels, which impair bone mineralization. Paradoxically, an important complication of XLH is mineralization of the enthesis (enthesopathy). Studies were undertaken to identify the cellular and molecular pathways important for normal post-natal enthesis maturation and to examine their role during the development of enthesopathy in mice with XLH (Hyp). The Achilles tendon entheses of Hyp mice demonstrate an expansion of hypertrophic-appearing chondrogenic cells by P14. Post-natally, cells in wild-type and Hyp entheses similarly descend from scleraxis- and Sox9-expressing progenitors; however, Hyp entheses exhibit an expansion of Sox9-expressing cells, and enhanced BMP and IHH signaling. These results support a role for enhanced BMP and IHH signaling in the development of enthesopathy in XLH.

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Citations

Aug 9, 2019·Pediatric Nephrology : Journal of the International Pediatric Nephrology Association·Marie-Eve RobinsonFrank Rauch
Oct 20, 2020·Frontiers in Cell and Developmental Biology·Carole-Anne Faraji-BelléeClaire Bardet
Nov 19, 2020·Journal of the Endocrine Society·Kathryn DahirJill H Simmons
Nov 27, 2020·Therapeutic Advances in Musculoskeletal Disease·Gavin Clunie, Nicole Horwood
Dec 10, 2021·Journal of Bone and Mineral Research : the Official Journal of the American Society for Bone and Mineral Research·Carlos R FerreiraDemetrios T Braddock

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