Switching from active vitamin D and phosphate supplementation to burosumab significantly corrects lower limb malalignment in pediatric X-linked hypophosphatemia
Main finding · Limbs with improved mFTAResult — go to the resultOdds ratio 4.38 95% CI 1.09–17.50Authors' conclusion
This study shows that switching to burosumab significantly improves lower limb malalignment in children with XLH over benefits conferred by active D/Pi, with early burosumab initiation providing the greatest benefit.
Study context
- Population
- Children with XLH enrolled in the XLH Disease Monitoring Program with a history of active D/Pi
- Exposure
- Switch from active D/Pi to burosumab vs remaining on active D/Pi
- Outcome
- Change in mechanical femoral tibial angle (mFTA), assessed per limb, Year 3 radiographs vs baseline
- Follow-up
- Year 3
Methodological detailsregistry-based, limb-level analysis, multivariate factor analysis
- Registry-based
- Limb-level analysis
- Multivariate factor analysis
Main finding
Limbs with improved mFTA
Odds ratio
4.38
95% CI 1.09–17.50
P = 0.0469
Burosumabvs Active D/Pi
OR 4.38 (95% CI 1.09–17.50); P = 0.0469
As reported in the abstract
Observational data: associations, not effects.
Further findings
Factors associated with greater change in mechanical femoral tibial angle (mFTA) Z-score (factor analysis of 24 attributes)
- Younger age at burosumab initiation
- P = 0.001
- Lower baseline height Z-score
- P = 0.006
Original publication
Switching from active vitamin D and phosphate supplementation to burosumab significantly corrects lower limb malalignment in pediatric X-linked hypophosphatemia. J Bone Miner Res. Published 13 June 2025. doi:10. 1093/ jbmr/ zjaf079
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