Markers and regulators of osteoblast and osteoclast activity in children with X-linked hypophosphatemia treated with burosumab
Authors' conclusion
In this real-world setting, pediatric XLH patients receiving burosumab treatment showed persisting mild hypophosphatemia and increased markers of osteoblast and osteoclast acitivity despite negative counter-regulation of osteoblasts by sclerostin and osteoclasts by OPG/RANKL.
Study context
- Population
- Burosumab-treated children with X-linked hypophosphatemia (XLH); median age 10.7 yr, median burosumab duration 2.2 yr; 18.7% on primary burosumab, 81.3% with prior phosphate and active vitamin D
- Participants
- 107 children
- Outcome
- z-scores of 7 markers and regulators of osteoblast and osteoclast activity (BAP, P1NP, CTX, TRAP5b, sRANKL, OPG, sclerostin)
Methodological detailsobservational, real-world
- Observational
- Real-world
Main finding
As reported in the abstract
Bone turnover markers and regulators (z-scores)
- Z-scores for osteoblast (BAP, P1NP) and osteoclast (CTX, TRAP5b) activity markers increased
- each p < .001
- Sclerostin z-score increased
- p < .001
- Standardized sRANKL reduced
- p < .05
- Osteoprotegerin (OPG) increased, sRANKL/OPG ratio reduced
- each p < .001
Observational data: associations, not effects.
Further findings
Height and BMI z-score change from baseline with burosumab
- Height and BMI improved from baseline
- ≈0.3 z-score
sRANKL, OPG and sRANKL/OPG ratio z-scores
- Standardized sRANKL reduced (p < .05)
- OPG increased (p < .001)
- sRANKL/OPG ratio reduced (p < .001)
Multivariate associations
- Serum sclerostin associated negatively with serum phosphate and positively with serum calcium and vitamin D sufficiency
- Primary burosumab treatment associated negatively with BAP
- Multivariate analysis
Parts of this Brief are withheld: the automatic checks could not confirm them against the abstract.
Original publication
Markers and regulators of osteoblast and osteoclast activity in children with X-linked hypophosphatemia treated with burosumab. J Bone Miner Res. Published 25 April 2026. doi:10.
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