Osteoporosis, bone & mineral

Switching from active vitamin D and phosphate supplementation to burosumab significantly corrects lower limb malalignment in pediatric X-linked hypophosphatemia

J Bone Miner ResPublished Observational study within the X-linked hypophosphatemia (XLH) Disease Monitoring Program

Main finding · Limbs with improved mFTAResult — go to the resultOdds ratio 4.38 95% CI 1.09–17.50

Authors' 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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