Burosumab vs Phosphate/Active Vitamin D in Pediatric X-Linked Hypophosphatemia: A Subgroup Analysis by Dose Level
Authors' conclusion
Prior phosphate or active vitamin D doses did not influence treatment response after switching to burosumab among children with XLH and active radiographic rickets. Switching from conventional therapy to burosumab improved rickets and serum ALP more than continuing either higher or lower doses of phosphate or active vitamin D.
Study context
- Population
- Children aged 1 to 12 years with X-linked hypophosphatemia (XLH)
- Participants
- 61 children
- Comparison
- Switching to burosumab vs continuing higher or lower doses of conventional therapy (active vitamin D and phosphate)
- Primary endpoint
- RGI-C for rickets by conventional therapy dose group
- Follow-up
- Week 64
Methodological detailsopen-label, phase 3
- Open-label
- Phase 3
Primary result
As reported in the abstract
HPi: burosumab vs conventional therapy
+1.72 vs +0.67
LPi: burosumab vs conventional therapy
+2.14 vs +1.08
HD: burosumab vs conventional therapy
+1.90 vs +0.94
LD: burosumab vs conventional therapy
+2.11 vs +1.06
Further findings
- Radiographic Global Impression of Change (RGI-C) for rickets by on-study dose group · week 64 — Burosumab
- +2.06
- RGI-C for rickets by on-study dose group — Conventional therapy HPi
- +1.03
- RGI-C for rickets by on-study dose group — Conventional therapy LPi
- +1.05
- RGI-C for rickets by on-study dose group — Conventional therapy HD
- +1.45
- RGI-C for rickets by on-study dose group — Conventional therapy LD
- +0.72
RGI-C for rickets by on-study dose group · week 64
- RGI-C higher with burosumab vs all on-study dose groups
Serum alkaline phosphatase (ALP)
- ALP decreased more with burosumab than conventional therapy, regardless of on-study phosphate and active vitamin D doses
Original publication
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