Serum Phosphorus as a Driver of Skeletal Morbidity in Fibrous Dysplasia
Authors' conclusion
Both frank hypophosphatemia and low-normophosphatemia are associated with increased FD-related complications. This supports FGF23-mediated hypophosphatemia as a driver of skeletal morbidity, which may impact a larger proportion of the FD/McCune-Albright syndrome population than previously recognized. These findings enable clinicians to identify at-risk patients and will inform development of prospective studies to determine optimal therapeutic targets.
Study context
- Population
- Subjects with fibrous dysplasia at a clinical research center with ≥1 fasting phosphorus level (age- and sex-adjusted Z-scores)
- Participants
- 240 subjects
- Comparison
- Frank hypophosphatemia and low-normophosphatemia vs high-normophosphatemia
- Frank hypophosphatemian = 48
- Low-normophosphatemian = 66
- High-normophosphatemian = 125reference
- Outcome
- Fractures, orthopedic surgeries and scoliosis
Methodological detailssingle clinical research center
- Single clinical research center
Main finding
As reported in the abstract
- Frank and low-normophosphatemia had increased fracture and surgery rates vs high-normophosphatemia
- Prevalence of moderate to severe scoliosis similarly higher in frank and low-normophosphatemia
Further findings
Fractures and surgeries in subanalysis matched for Skeletal Burden Score ≥35
- Fracture and surgery rates remained higher with frank hypophosphatemia
- Matched for Skeletal Burden Score ≥35
Original publication
Serum Phosphorus as a Driver of Skeletal Morbidity in Fibrous Dysplasia. J Clin Endocrinol Metab. Published 17 November 2023. doi:10.
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